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1. Terzic A, Bode B, Gratz KW, Stoeckli SJ: Prognostic factors for the malignant triton tumor of the head and neck. Head Neck; 2009 May;31(5):679-88
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Prognostic factors for the malignant triton tumor of the head and neck.
  • BACKGROUND: Malignant triton tumors are rare neoplasias consisting of a malignant peripheral nerve sheath tumor with additional rhabdomyoblastic differentiation.
  • These tumors are highly aggressive and prognosis is poor.
  • METHODS: From 1993 to 2005, 7 patients with a malignant triton tumor of the head and neck were treated at our institution.
  • RESULTS: Patients with primary tumors involving the nose and paranasal sinuses have better, patients involving the neck a poor prognosis.
  • Additional radiation or chemotherapy show little effect.
  • CONCLUSION: Location of the primary tumor is a key factor for prognosis.
  • Complete surgical removal is the only treatment associated with survival.
  • [MeSH-major] Head and Neck Neoplasms / mortality. Nerve Sheath Neoplasms / mortality

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  • (PMID = 19283843.001).
  • [ISSN] 1097-0347
  • [Journal-full-title] Head & neck
  • [ISO-abbreviation] Head Neck
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 47
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2. Neville H, Corpron C, Blakely ML, Andrassy R: Pediatric neurofibrosarcoma. J Pediatr Surg; 2003 Mar;38(3):343-6; discussion 343-6
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  • [Title] Pediatric neurofibrosarcoma.
  • BACKGROUND/PURPOSE: Neurofibrosarcoma is rare in children, and the natural history and prognostic factors are not well described.
  • METHODS: The charts of children with neurofibrosarcoma were reviewed retrospectively.
  • RESULTS: From 1944 to 2001, 38 patients under the age of 21 were diagnosed with neurofibrosarcoma.
  • The average age at diagnosis was 13.8 years (range, 3 to 19.9 years).
  • The tumor site was as follows: extremity, 19 patients; trunk, 9 patients; head and neck, 7 patients; and retroperitoneum, 3 patients.
  • The average tumor size was 10 cm.
  • Thirty-two patients achieved a complete response, 2 a partial response, and 4 progressed while on therapy.
  • Of the 15 patients with a distant relapse, 73% (11) relapsed in the lung.
  • Nine patients were treated with chemotherapy, 9 with radiation, and 9 with both chemotherapy and radiation.
  • Outcome was not significantly affected by gender, presence of neurofibromatosis, site, margin, or use of adjuvant therapy.
  • CONCLUSION: Neurofibrosarcoma remains a rare disease in children with insufficient contemporary numbers to assess efficacy of therapy.
  • Prognosis remains poor with a high incidence of relapse, particularly in the lungs, suggesting that more aggressive therapies to control both local and distant relapses are needed.
  • [MeSH-major] Neurofibrosarcoma / epidemiology. Soft Tissue Neoplasms / epidemiology
  • [MeSH-minor] Adolescent. Adult. Chemotherapy, Adjuvant. Child. Child, Preschool. Combined Modality Therapy. Female. Genetic Predisposition to Disease. Humans. Lung Neoplasms / epidemiology. Lung Neoplasms / secondary. Male. Neoplasms, Second Primary / epidemiology. Neurofibromatoses / epidemiology. Prognosis. Radiotherapy, Adjuvant. Retrospective Studies. Survival Rate. Texas / epidemiology. Treatment Outcome

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  • [Copyright] Copyright 2003, Elsevier Science (USA). All rights reserved.
  • (PMID = 12632346.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 18
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3. Dilworth JT, Wojtkowiak JW, Mathieu P, Tainsky MA, Reiners JJ Jr, Mattingly RR, Hancock CN: Suppression of proliferation of two independent NF1 malignant peripheral nerve sheath tumor cell lines by the pan-ErbB inhibitor CI-1033. Cancer Biol Ther; 2008 Dec;7(12):1938-46
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  • [Title] Suppression of proliferation of two independent NF1 malignant peripheral nerve sheath tumor cell lines by the pan-ErbB inhibitor CI-1033.
  • Neurofibromatosis Type 1 (NF1) is characterized by the abnormal proliferation of neuroectodermal tissues and the development of certain tumors, particularly neurofibromas, which may progress into malignant peripheral nerve sheath tumors (MPNSTs).
  • Effective pharmacological therapy for the treatment of NF1 tumors is currently unavailable and the prognosis for patients with MPNSTs is poor.
  • Loss of neurofibromin correlates with increased expression of the epidermal growth factor receptor (EGFR) and ErbB2 tyrosine kinases and these kinases have been shown to promote NF1 tumor-associated pathologies in vivo.
  • We show here that while NF1 MPNST cells have higher EGFR expression levels and are more sensitive to EGF when compared to a non-NF1 MPNST cell line, the ability of the EGFR inhibitor gefitinib to selectively inhibit NF1 MPNST cell proliferation is marginal.
  • We also show that NF1 MPNST proliferation correlates with activated ErbB2 and can be suppressed by nanomolar concentrations of the pan-ErbB inhibitor CI-1033 (canertinib).
  • Consequently, targeting both EGFR and ErbB2 may prove an effective strategy for suppressing NF1 MPNST tumor growth in vivo.

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  • (PMID = 18927496.001).
  • [ISSN] 1555-8576
  • [Journal-full-title] Cancer biology & therapy
  • [ISO-abbreviation] Cancer Biol. Ther.
  • [Language] ENG
  • [Grant] United States / NIEHS NIH HHS / ES / T32 ES012163; United States / NIEHS NIH HHS / ES / P30 ES06639; United States / NIEHS NIH HHS / ES / P30 ES006639; United States / NCI NIH HHS / CA / P30 CA022453; United States / NCI NIH HHS / CA / P30 CA22453
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Morpholines; 0 / Quinazolines; C78W1K5ASF / Canertinib; EC 2.7.10.1 / Protein-Tyrosine Kinases; EC 2.7.10.1 / Receptor, Epidermal Growth Factor; EC 2.7.10.1 / Receptor, ErbB-2; S65743JHBS / gefitinib
  • [Other-IDs] NLM/ NIHMS525367; NLM/ PMC3923431
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4. Satoh M, Komori K, Shin M, Takada T, Honda M, Fujioka H, Tsujimoto M: [Retroperitoneal malignant peripheral nerve sheath tumor (MPNST) complicated with von Recklinghausen's disease: a case report]. Hinyokika Kiyo; 2004 Jun;50(6):417-20
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  • [Title] [Retroperitoneal malignant peripheral nerve sheath tumor (MPNST) complicated with von Recklinghausen's disease: a case report].
  • A retroperitoneal malignant peripheral nerve sheath tumor (MPNST) in a patient with von Recklinghausen's disease is reported.
  • A 55-year-old woman was admitted with a left side abdominal mass.
  • Computed tomography (CT) and magnetic resonance imaging (MRI) revealed a 9 x 9 cm retroperitoneal mass.
  • Two other tumors were also found.
  • The retroperitoneal tumor was resected en bloc.
  • The tumor was a solid yellow mass.
  • Macroscopically it has a pseudocapsule of fibrous tissue, weighed 1,120 g and measured 9 x 9 x 15 cm.
  • The histopathological diagnosis was malignant peripheral nerve sheath tumor (MPNST).
  • Since the responsiveness of these tumors to chemotherapy and radiation therapy is poor, we did not administer adjuvant therapy.
  • [MeSH-major] Nerve Sheath Neoplasms / etiology. Neurofibromatosis 1 / complications. Retroperitoneal Neoplasms / etiology

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  • (PMID = 15293741.001).
  • [ISSN] 0018-1994
  • [Journal-full-title] Hinyokika kiyo. Acta urologica Japonica
  • [ISO-abbreviation] Hinyokika Kiyo
  • [Language] jpn
  • [Publication-type] Case Reports; English Abstract; Journal Article; Review
  • [Publication-country] Japan
  • [Number-of-references] 7
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5. Kopp HG, Kanz L, Hartmann JT: Hypersensitivity pneumonitis associated with the use of trofosfamide. Anticancer Drugs; 2004 Jul;15(6):603-4
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  • Oral continuous low-dose therapy has been the most widely used schedule.
  • Higher grades of nephrotoxicity or neurotoxicity--side-effects that typically limit the use of ifosfamide-have not been reported with low-dose continuous trofosfamide treatment.
  • We report herein a case of a 83-year-old female patient with a disseminated malignant peripheral nerve sheath tumor treated with trofosfamide developing pulmonal toxicity.
  • To our knowledge, this is the first reported case of exogenous allergic alveolitis after exposure to this drug.
  • [MeSH-minor] Administration, Oral. Aged. Antineoplastic Combined Chemotherapy Protocols. Cystitis / chemically induced. Cystitis / complications. Drug Administration Schedule. Female. Femur / pathology. Germany. Humans. Ifosfamide / adverse effects. Ifosfamide / metabolism. Nausea / chemically induced. Nausea / complications. Neoplasm Metastasis / diagnosis. Neoplasm Metastasis / drug therapy. Neoplasm Metastasis / pathology. Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / drug therapy. Nerve Sheath Neoplasms / pathology. Phosphoramide Mustards / administration & dosage. Phosphoramide Mustards / adverse effects. Phosphoramide Mustards / metabolism. Prednisolone / administration & dosage. Prednisolone / therapeutic use. Pulmonary Fibrosis / complications. Pulmonary Fibrosis / diagnosis. Pulmonary Fibrosis / drug therapy. Vomiting / chemically induced. Vomiting / complications

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  • (PMID = 15205604.001).
  • [ISSN] 0959-4973
  • [Journal-full-title] Anti-cancer drugs
  • [ISO-abbreviation] Anticancer Drugs
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Phosphoramide Mustards; 8N3DW7272P / Cyclophosphamide; 9PHQ9Y1OLM / Prednisolone; H64JRU6GJ0 / trofosfamide; UM20QQM95Y / Ifosfamide
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6. Katz D, Lazar A, Lev D: Malignant peripheral nerve sheath tumour (MPNST): the clinical implications of cellular signalling pathways. Expert Rev Mol Med; 2009;11:e30
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Malignant peripheral nerve sheath tumour (MPNST): the clinical implications of cellular signalling pathways.
  • Malignant peripheral nerve sheath tumour (MPNST) is a rare malignancy accounting for 3-10% of all soft tissue sarcomas.
  • Most MPNSTs arise in association with peripheral nerves or deep neurofibromas and may originate from neural crest cells, although the specific cell of origin is uncertain.
  • Approximately half of MPNSTs occur in the setting of neurofibromatosis type 1 (NF1), an autosomal dominant disorder with an incidence of approximately one in 3500 persons; the remainder of MPNSTs develop sporadically.
  • In addition to a variety of clinical manifestations, approximately 8-13% of NF1 patients develop MPNSTs, which are the leading cause of NF1-related mortality.
  • Surgical resection is the mainstay of MPNST clinical management.
  • However, because of invasive growth, propensity to metastasise, and limited sensitivity to chemotherapy and radiation, MPNST has a guarded to poor prognosis.
  • Five-year survival rates of only 20-50% indicate an urgent need for improved therapeutic approaches.
  • Recent work in this field has identified several altered intracellular signal transduction cascades and deregulated tyrosine kinase receptors, posing the possibility of personalised, targeted therapeutics.
  • However, expanded knowledge of MPNST molecular pathobiology will be needed to meaningfully apply such approaches for the benefit of afflicted patients.
  • [MeSH-major] Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / metabolism. Neurofibromin 1 / metabolism. Protein Kinases / metabolism. Proto-Oncogene Proteins p21(ras) / metabolism

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  • (PMID = 19835664.001).
  • [ISSN] 1462-3994
  • [Journal-full-title] Expert reviews in molecular medicine
  • [ISO-abbreviation] Expert Rev Mol Med
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Neurofibromin 1; EC 2.7.- / Protein Kinases; EC 2.7.1.1 / MTOR protein, human; EC 2.7.1.1 / TOR Serine-Threonine Kinases; EC 2.7.10.1 / EGFR protein, human; EC 2.7.10.1 / Receptor, Epidermal Growth Factor; EC 2.7.10.1 / Receptors, Platelet-Derived Growth Factor; EC 2.7.10.1 / Receptors, Vascular Endothelial Growth Factor; EC 3.6.5.2 / Proto-Oncogene Proteins p21(ras)
  • [Number-of-references] 135
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7. Lejeune FJ, Pujol N, Liénard D, Mosimann F, Raffoul W, Genton A, Guillou L, Landry M, Chassot PG, Chiolero R, Bischof-Delaloye A, Leyvraz S, Mirimanoff RO, Bejkos D, Leyvraz PF: Limb salvage by neoadjuvant isolated perfusion with TNFalpha and melphalan for non-resectable soft tissue sarcoma of the extremities. Eur J Surg Oncol; 2000 Nov;26(7):669-78
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  • [Title] Limb salvage by neoadjuvant isolated perfusion with TNFalpha and melphalan for non-resectable soft tissue sarcoma of the extremities.
  • AIMS: Patients with non-resectable soft tissue sarcomas of the extremities do not live longer if they are treated by amputation or disarticulation.
  • In order to avoid major amputations, we tested isolated limb perfusion (ILP) with tumour necrosis factor alpha (TNF)+melphalan+/-interferon-gamma (IFN) as a pre-operative, neoadjuvant limb salvage treatment.
  • METHODS: Twenty-two patients were included (six men and 16 women; three upper limb and 19 lower limb tumours).
  • Thirteen cases were recurrent or progressive after previous therapy; five tumours had a diameter >/=20 cm, and four were multiple or regionally metastatic.
  • There were six malignant fibrous histiocytomas, five liposarcomas, four malignant peripheral nerve sheath tumours, three rhabdomyosarcomas, two leiomyosarcomas, one recurrent extraskeletal osteosarcoma and one angiosarcoma.
  • Three patients had a minimal or no response and the tumour progressed in one case.
  • All patients had fever for 24 hours but only one developed a reversible grade 3 distributive shock syndrome with no sequelae.
  • Seventeen patients (77%) underwent limb-sparing resection of the tumour remnants after a median time of 3.4 months: 10 resections were intracompartmental and seven extracompartmental.
  • Adjuvant chemotherapy was given to eight patients and radiotherapy to six.
  • The median disease free and overall survival times have been >12.5 and 18.7 months respectively: this is similar to the outcome after primary amputations for similar cases.
  • CONCLUSION: ILP with TNF and chemotherapy is an efficient limb sparing neoadjuvant therapy for a priori non-resectable limb soft tissue sarcomas.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Leg / surgery. Sarcoma / drug therapy. Sarcoma / surgery. Soft Tissue Neoplasms / drug therapy. Soft Tissue Neoplasms / surgery
  • [MeSH-minor] Adult. Aged. Chemotherapy, Adjuvant. Chemotherapy, Cancer, Regional Perfusion. Disease-Free Survival. Doxorubicin / administration & dosage. Female. Humans. Ifosfamide / administration & dosage. Interferon-gamma / administration & dosage. Interferon-gamma / adverse effects. Male. Melphalan / administration & dosage. Melphalan / adverse effects. Middle Aged. Neoadjuvant Therapy. Neoplasm Recurrence, Local / surgery. Radiotherapy, Adjuvant. Salvage Therapy. Survival Analysis. Tumor Necrosis Factor-alpha / administration & dosage. Tumor Necrosis Factor-alpha / adverse effects

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  • [Copyright] Copyright 2000 Harcourt Publishers Ltd.
  • (PMID = 11078614.001).
  • [ISSN] 0748-7983
  • [Journal-full-title] European journal of surgical oncology : the journal of the European Society of Surgical Oncology and the British Association of Surgical Oncology
  • [ISO-abbreviation] Eur J Surg Oncol
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article
  • [Publication-country] ENGLAND
  • [Chemical-registry-number] 0 / Tumor Necrosis Factor-alpha; 80168379AG / Doxorubicin; 82115-62-6 / Interferon-gamma; Q41OR9510P / Melphalan; UM20QQM95Y / Ifosfamide
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8. Landy H, Feun L, Markoe A, Patchen S, Bruce J, Marcus J, Levi A: Extended remission of a recurrent median nerve malignant peripheral nerve sheath tumor after multimodal treatment. Case report. J Neurosurg; 2005 Oct;103(4):760-3
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Extended remission of a recurrent median nerve malignant peripheral nerve sheath tumor after multimodal treatment. Case report.
  • Malignant peripheral nerve sheath tumors (MPNSTs) are difficult to control despite aggressive treatment.
  • In this report the authors describe the treatment and follow-up review of a patient with neurofibromatosis Type I who harbored a recurrent median nerve MPNST.
  • The man underwent preoperative intraarterial and intravenous chemotherapy followed by additional surgery for gross-total removal and postoperative radiotherapy.
  • Two courses of preoperative intraarterial cisplatin and intravenous Adriamycin produced significant tumor shrinkage.
  • Gross-total removal of the remaining tumor without amputation of the arm was followed by fractionated radiotherapy (total minimum tumor dose 6485 cGy, maximal dose 6575 cGy).
  • The patient is alive 9.5 years after treatment without evidence of tumor recurrence and with only focal median nerve functional deficits.
  • A review of the patient's treatment is warranted to provide a description of a regimen that may be useful in the treatment of similar patients in the future.
  • [MeSH-major] Neoplasm Recurrence, Local / surgery. Nerve Sheath Neoplasms / surgery. Neurofibromatosis 1 / surgery
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Cisplatin / administration & dosage. Combined Modality Therapy. Doxorubicin / administration & dosage. Humans. Male. Treatment Outcome


9. Häcker FM, von Schweinitz D, Gambazzi F: The relevance of surgical therapy for bilateral and/or multiple pulmonary metastases in children. Eur J Pediatr Surg; 2007 Apr;17(2):84-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] The relevance of surgical therapy for bilateral and/or multiple pulmonary metastases in children.
  • PURPOSE: Pulmonary surgery is frequently used for the treatment of metastases in children with various malignant diseases.
  • The benefit of an aggressive surgical treatment in children with bilateral and/or multiple pulmonary metastases is still discussed controversially.
  • The primary malignancies were osteosarcoma (n = 4), hepatoblastoma (n = 3), malignant peripheral nerve sheath tumor (n = 1), adrenocortical carcinoma (n = 1) and alveolar rhabdomyosarcoma (n = 1).
  • Preoperative induction chemotherapy with tumor regression and a subsequent decrease in the size and number of pulmonary metastases was mandatory for the surgery of metastases.
  • RESULTS: Standardized bilateral thoracotomy was performed in 4 patients in 1 operation (in 1 patient combined with a hemihepatectomy), and in 3 patients, in 2 operations on different days.
  • CONCLUSION: Complete surgical resection of pulmonary metastases after response to induction chemotherapy may increase survival in carefully selected children, even in cases with multiple and recurrent metastatic disease.

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  • (PMID = 17503299.001).
  • [ISSN] 0939-7248
  • [Journal-full-title] European journal of pediatric surgery : official journal of Austrian Association of Pediatric Surgery ... [et al] = Zeitschrift für Kinderchirurgie
  • [ISO-abbreviation] Eur J Pediatr Surg
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
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10. Bhola P, Banerjee S, Mukherjee J, Balasubramanium A, Arun V, Karim Z, Burrell K, Croul S, Gutmann DH, Guha A: Preclinical in vivo evaluation of rapamycin in human malignant peripheral nerve sheath explant xenograft. Int J Cancer; 2010 Jan 15;126(2):563-71
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  • [Title] Preclinical in vivo evaluation of rapamycin in human malignant peripheral nerve sheath explant xenograft.
  • Neurofibromatosis type 1 (NF1) patients are prone to the development of malignant tumors, the most common being Malignant Peripheral Nerve Sheath Tumor (MPNST).
  • NF1-MPNST patients have an overall poor survival due to systemic metastasis.
  • Currently, the management of MPNSTs includes surgery and radiation; however, conventional chemotherapy is not very effective, underscoring the need for effective biologically-targeted therapies.
  • Recently, the NF1 gene product, neurofibromin, was shown to negatively regulate the phosphoinositide-3-kinase (PI3K)/Protein Kinase-B (Akt)/mammalian Target Of Rapamycin (mTOR) pathway, with loss of neurofibromin expression in established human MPNST cell lines associated with high levels of mTOR activity.
  • We developed and characterized a human NF1-MPNST explant grown subcutaneously in NOD-SCID mice, to evaluate the effect of the mTOR inhibitor rapamycin.
  • We demonstrate that rapamycin significantly inhibited human NF1-MPNST mTOR pathway activation and explant growth in vivo at doses as low as 1.0 mg/kg/day, without systemic toxicities.
  • While rapamycin was effective at reducing NF1-MPNST proliferation and angiogenesis, with decreased CyclinD1 and VEGF respectively, there was no increase in tumor apoptosis.
  • This study demonstrates the therapeutic potential and limitations of rapamycin in NF1-associated, and likely sporadic, MPNSTs.
  • [MeSH-major] Neurofibromatosis 1 / drug therapy. Peripheral Nervous System Neoplasms / drug therapy. Sirolimus / pharmacology. Xenograft Model Antitumor Assays
  • [MeSH-minor] Animals. Antibiotics, Antineoplastic / pharmacology. Apoptosis / drug effects. Blotting, Western. Cyclin D1 / metabolism. Dose-Response Relationship, Drug. Drug Evaluation, Preclinical. Humans. Immunohistochemistry. Male. Mice. Mice, Inbred NOD. Mice, SCID. Protein Kinases / metabolism. Proto-Oncogene Proteins c-akt / metabolism. Ribosomal Protein S6 Kinases / metabolism. Signal Transduction / drug effects. TOR Serine-Threonine Kinases. Tumor Burden / drug effects. Vascular Endothelial Growth Factor A / metabolism. Young Adult

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  • (PMID = 19634141.001).
  • [ISSN] 1097-0215
  • [Journal-full-title] International journal of cancer
  • [ISO-abbreviation] Int. J. Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antibiotics, Antineoplastic; 0 / Vascular Endothelial Growth Factor A; 136601-57-5 / Cyclin D1; EC 2.7.- / Protein Kinases; EC 2.7.1.1 / MTOR protein, human; EC 2.7.1.1 / TOR Serine-Threonine Kinases; EC 2.7.1.1 / mTOR protein, mouse; EC 2.7.11.1 / Proto-Oncogene Proteins c-akt; EC 2.7.11.1 / Ribosomal Protein S6 Kinases; W36ZG6FT64 / Sirolimus
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11. Vander Salm TJ: Unusual primary tumors of the heart. Semin Thorac Cardiovasc Surg; 2000 Apr;12(2):89-100
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  • [Title] Unusual primary tumors of the heart.
  • Primary tumors of the heart, with the exception of atrial myxomas, occur rarely; tumors metastatic to or directly invasive of the heart are far more common.
  • About 75% of primary tumors are benign, and 75% of these are atrial myxomas.
  • The benign tumors include rhabdomyomas, fibromas, papillary fibroelastomas, hemangiomas, pericardial cysts, lipomas, hamartomas, teratomas, mesotheliomas, and paragangliomas or pheochromocytomas.
  • The last 3 may also be malignant.
  • The malignant tumors consist of various sarcomas: myxosarcoma, liposarcoma, angiosarcoma, fibrosarcoma, leiomyosarcoma, osteosarcoma, synovial sarcoma, rhabdomyosarcoma, undifferentiated sarcoma, reticulum cell sarcoma, neurofibrosarcoma, and malignant fibrous histiocytoma.
  • Cardiac tumors produce a large variety of symptoms through any of 4 mechanisms.
  • Bits of tumor can embolize, causing systemic deficits when the tumors are on the left side of the heart.
  • Finally, the tumors may cause systemic or constitutional symptoms.
  • Some tumors, of course, produce no symptoms and become evident as incidental findings.
  • The most useful diagnostic tool is the echocardiogram, which in almost all cases precisely locates the tumor and defines its extent.
  • The echocardiographic appearance may also allow quite accurate prediction of the tumor type and whether it is malignant or benign.
  • Magnetic resonance imaging serves as the next most important test where the density of T1 and T2 images may allow tumor cell type identification.
  • With few exceptions, these tumors require operative excision.
  • Most benign tumors can be resected completely; a few, because of their large size, cannot be, and only tumor debulking may be possible.
  • Many of the malignant tumors cannot be resected completely, either because of the extent of local spread and invasion or because of the frequent distant metastases.
  • The long-term results for resected benign tumors are excellent; the long-term results for sarcomas are very poor, and there are few survivors.
  • For patients with unresectable sarcomas, radiation and chemotherapy may be used, but without great expectation of successful results.
  • [MeSH-major] Heart Neoplasms / diagnosis. Heart Neoplasms / surgery
  • [MeSH-minor] Fibroma / diagnosis. Fibroma / surgery. Hamartoma / diagnosis. Hamartoma / surgery. Heart Septum / pathology. Heart Transplantation. Hemangioma / diagnosis. Hemangioma / surgery. Humans. Hypertrophy. Mesothelioma / diagnosis. Mesothelioma / surgery. Pheochromocytoma / diagnosis. Pheochromocytoma / surgery. Prognosis. Rhabdomyoma / diagnosis. Rhabdomyoma / surgery. Teratoma / diagnosis. Teratoma / surgery

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  • (PMID = 10807431.001).
  • [ISSN] 1043-0679
  • [Journal-full-title] Seminars in thoracic and cardiovascular surgery
  • [ISO-abbreviation] Semin. Thorac. Cardiovasc. Surg.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] UNITED STATES
  • [Number-of-references] 69
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12. Hatori M, Hosaka M, Watanabe M, Moriya T, Sasano H, Kokubun S: Osteosarcoma in a patient with neurofibromatosis type 1: a case report and review of the literature. Tohoku J Exp Med; 2006 Apr;208(4):343-8
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  • [Title] Osteosarcoma in a patient with neurofibromatosis type 1: a case report and review of the literature.
  • Neurofibromatosis type 1 (NF1) or von Recklinghausen's disease is a genetic disease generally characterized by café-au-lait spots and neurofibromas.
  • Malignant tumors of the nervous system, such as malignant schwannomas, gliomas, or astrocytomas, have been well known to coexist with neurofibromatosis.
  • However, occurrence of malignant tumors unrelated to the nervous system is rare.
  • We report an unusual case of a 29-year-old NF1 female suffering from malignant peripheral nerve sheath tumor (MPNST) that eventually developed osteosarcoma in the proximal femur.
  • Osteosarcoma is the most common high-grade malignant bone tumor in which the neoplastic cells produce osteoid.
  • At 23 and 24 years old, she underwent excision of MPNST in the left posterior thigh.
  • The patient underwent postoperative chemotherapy.
  • Computed tomography demonstrated cortical bone destruction in the left proximal femur where MPNST occurred.
  • Magnetic resonance imaging revealed extraskeletal growth of the tumor.
  • The removed tumor was composed of highly anaplastic cells.
  • Lace-like irregular osteoid formation was observed among the tumor cells.
  • MPNST component was totally absent.
  • The tumor was diagnosed as osteoblastic type osteosarcoma.
  • The correlation between the histogenesis of osteosarcoma and the genetic abnormality in NF1 patients has not been elucidated, but the finding of osteosarcomatous transformation in this case suggests the divergent cellular differentiation to mesenchymal malignant tumors of neuroectodermal tissue in NF1 patients.
  • [MeSH-minor] Adult. Cell Transformation, Neoplastic. Combined Modality Therapy. Fatal Outcome. Female. Femur / pathology. Femur / radiography. Humans. Magnetic Resonance Imaging. Nerve Sheath Neoplasms / pathology. Nerve Sheath Neoplasms / therapy


13. Jeyaretna DS, Rabkin SD, Martuza RL: Oncolytic herpes simplex virus therapy for peripheral nerve tumors. Neurosurg Focus; 2007;22(6):E4
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  • [Title] Oncolytic herpes simplex virus therapy for peripheral nerve tumors.
  • Oncolytic viruses are one of many emerging cancer therapies.
  • The surgical management of peripheral nerve tumors carries an inherent risk of damaging the nerves involved and so the search for novel therapies with reduced risk of morbidity continues.
  • In this review the authors discuss the use of oncolytic herpes simplex virus (HSV) in the treatment of peripheral nerve tumors.
  • Herpes simplex virus has a number of characteristics that make it a useful oncolytic vector, including its large, sequenced genome that can accommodate multiple transgenes, its lack of insertional mutagenesis, its ability to infect a wide array of cell types in various species, and the availability of well-established antiviral therapies to treat it.
  • The efficacy of oncolytic HSV therapy against schwannomas and malignant peripheral nerve sheath tumors has been studied in multiple experimental models both in vitro and in vivo.
  • The virus utilizes cell pathways unique to tumors to enhance its oncolytic efficacy, preferentially and effectively targeting and destroying peripheral nerve tumor cells without harming normal cells.
  • This effect is augmented by transgenes expressing antiangiogenic factors, such as dominant-negative fibroblast growth factor receptor and platelet factor 4, and displays synergy with chemotherapy.
  • In addition, new animal models have been developed to test the efficacy of oncolytic HSV therapy in peripheral nerve tumors.
  • [MeSH-major] Oncolytic Virotherapy / methods. Peripheral Nervous System Neoplasms / therapy. Simplexvirus / genetics
  • [MeSH-minor] Animals. Genetic Vectors / therapeutic use. Humans. Oncolytic Viruses / genetics

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  • (PMID = 17613221.001).
  • [ISSN] 1092-0684
  • [Journal-full-title] Neurosurgical focus
  • [ISO-abbreviation] Neurosurg Focus
  • [Language] eng
  • [Grant] United States / NINDS NIH HHS / NS / R01 NS032677
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, U.S. Gov't, Non-P.H.S.; Review
  • [Publication-country] United States
  • [Number-of-references] 37
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14. Gachiani J, Kim D, Nelson A, Kline D: Surgical management of malignant peripheral nerve sheath tumors. Neurosurg Focus; 2007;22(6):E13
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  • [Title] Surgical management of malignant peripheral nerve sheath tumors.
  • OBJECT: The aim of this study was to describe the presentation of patients harboring soft tissue sarcomas involving the nerves, most of which were malignant peripheral nerve sheath tumors (MPNSTs), and provide an algorithm for their treatment.
  • METHODS: The authors retrospectively analyzed data on 43 surgically treated soft tissue sarcomas involving the nerves, 34 (79%) of which were MPNSTs.
  • Tumor classifications are presented, together with patient numbers, locations of MPNSTs, surgical techniques, and adjunctive treatments.
  • RESULTS: The 34 MPNSTs were surgically treated during a period of 40 years.
  • Most of these lesions (19 MPNSTs [56%]) were located in the brachial plexus, whereas the rest were located on other major nerves.
  • Neurofibromatosis Type 1-associated tumors (12 lesions) represented 35% of the total number of MPNSTs.
  • CONCLUSIONS: Malignant peripheral nerve sheath tumors are rare.
  • The role of chemotherapy is still being defined.
  • [MeSH-major] Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / surgery
  • [MeSH-minor] Adolescent. Adult. Aged. Aged, 80 and over. Antineoplastic Agents / therapeutic use. Diagnosis, Differential. Disease Management. Humans. Middle Aged. Retrospective Studies

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  • (PMID = 17613204.001).
  • [ISSN] 1092-0684
  • [Journal-full-title] Neurosurgical focus
  • [ISO-abbreviation] Neurosurg Focus
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents
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15. Antoniello L, Cecchetto G, Carli M, Dall'Igna P, Bisogno G, Lo Piccolo R, Gigante C, Zanetti I, Guglielmi M: [Role of mutilating surgery in the treatment of non-chemosensitive pediatric soft tissue sarcomas. Experience of the Italian Cooperative Group Studies RMS-79 and RMS-88]. Pediatr Med Chir; 2003 Jul-Aug;25(4):255-60
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  • [Title] [Role of mutilating surgery in the treatment of non-chemosensitive pediatric soft tissue sarcomas. Experience of the Italian Cooperative Group Studies RMS-79 and RMS-88].
  • [Transliterated title] Ruolo della chirurgia mutilante nel trattamento dei sarcomi delle parti molli non chemiosensibili in età pediatrica. Esperienza degli studi Cooperativi Italiani RMS-79 ed RMS-88.
  • Aim of the study was to evaluate the role of mutilating surgery in the patients with non chemosensitive soft tissue sarcomas (STS) registered in the Italian Studies.
  • HISTOLOGY: fibrosarcoma 29, Malignant Perpheral Nerve Sheath Tumors (MPNST) 40, malignant fibrous histiocytoma 5, hemangiopericytoma 6, leiomyosarcoma 4, others 20, STS nos 10.
  • Twelve out of 114 pts (7%), 5/33 (14%) in the first study and 7/81 (8%) in the second, underwent mutilating surgery: 8 pts (of whom 3 were < 2 y of age) had a fibrosarcoma and 4 a MPNST.
  • The mutilating procedure was carried out at diagnosis in 6 cases (4 in RMS-79 and 2 in RMS-and 88) and achieved radicality in 5/6 cases.
  • It was performed after ineffective chemotherapy (CT) in 5 pts (1 in RMS-79 and 4 in RMS-88).
  • OUTCOME: At present 6/12 pts, 5 with fibrosarcoma and 1 with MPNST, are alive with no evidence of disease (NED), 4 of the first and 2 of the second study.
  • Of the 5 Gr. I patients, 4 are alive (NED) and 1 died of 2nd tumor; 1 Gr.
  • III pts 1 is alive NED and 4 died (3 of metastatic spread and 1 of 2nd tumor); the pt amputated after repeated local relapses (Gr.
  • CONCLUSIONS: In the RMS-79 study the mutilations were frequent and were performed at diagnosis in several cases; this trend decreased in the 2nd study in which chemotherapy was attempted in most of the patients.
  • Only fibrosarcomas and MPNST probably requires a more aggressive surgical behaviour.
  • [MeSH-major] Sarcoma / surgery. Soft Tissue Neoplasms / surgery

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  • (PMID = 15070267.001).
  • [ISSN] 0391-5387
  • [Journal-full-title] La Pediatria medica e chirurgica : Medical and surgical pediatrics
  • [ISO-abbreviation] Pediatr Med Chir
  • [Language] ita
  • [Publication-type] English Abstract; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Italy
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16. López Alvarez F, Llorente Pendás JL, Coca Pelaz A, Fernández García MS, Cuello Bueno G, Suárez Nieto C: Malignant triton tumor of the infratemporal fossa. J Craniofac Surg; 2009 Jul;20(4):1282-6
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  • [Title] Malignant triton tumor of the infratemporal fossa.
  • Malignant triton tumor is a very aggressive type of sarcoma that comprises rhabdomyoblasts and malignant Schwann cells.
  • It is a different entity from malignant schwannoma, characterized by their aggressiveness and poor prognosis.
  • Head and neck location is frequent, and early diagnosis and complete resection followed by radiation therapy is important for long-term survival.
  • However, the therapeutic plan should be individualized, taking into account the location and size of the primary tumor.
  • The use of adjuvant chemotherapy and molecular therapies should be considered in the treatment of these tumors.
  • We report an unusual presentation of a malignant triton tumor located in the infratemporal fossa, describing its clinical and pathologic features, and we try to update the knowledge in the management of these tumors, including the use of molecular therapies.
  • [MeSH-major] Hamartoma / diagnosis. Skull Base Neoplasms / diagnosis
  • [MeSH-minor] Biopsy. Combined Modality Therapy. Diagnosis, Differential. Endoscopy. Fatal Outcome. Female. Humans. Tomography, X-Ray Computed. Young Adult

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  • (PMID = 19625850.001).
  • [ISSN] 1536-3732
  • [Journal-full-title] The Journal of craniofacial surgery
  • [ISO-abbreviation] J Craniofac Surg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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17. Kolarov V, Stanić J, Eri Z, Zvezdin B, Kojičić M, Hromis S: Intrathoracic malignant peripheral nerve sheath tumor with poor outcome: a case report. Bosn J Basic Med Sci; 2010 Nov;10(4):328-30
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  • [Title] Intrathoracic malignant peripheral nerve sheath tumor with poor outcome: a case report.
  • We report a case of intrathoracic malignant peripheral nerve sheath tumor in a 65-year old woman revealed after a few-month history of progressive dyspnea, appetite and body mass loss.
  • The chest magnetic resonance (MR) examination revealed the presence of a large tumor occupying the mediastinum and a major portion of the right hemithorax.
  • The diagnostic tumor sample was obtained by parasternal biopsy in local anesthesia.
  • The surgical resection of the tumor could not be performed due to its excessive size, intrathoracic involvement and bad respiratory reserves of a patient.
  • The chemotherapy and irradiation were performed as palliative measures.
  • The lethal outcome appeared 10 months after the diagnosis was established.
  • [MeSH-major] Mediastinal Neoplasms / diagnosis. Mediastinal Neoplasms / therapy. Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / therapy. Peripheral Nerves / pathology
  • [MeSH-minor] Aged. Anesthesia / methods. Biopsy / methods. Fatal Outcome. Female. Humans. Lung / pathology. Magnetic Resonance Imaging / methods. Mediastinum / pathology. Pulmonary Disease, Chronic Obstructive / complications. Thorax / pathology. Treatment Outcome

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  • [Journal-full-title] Bosnian journal of basic medical sciences
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18. Johansson G, Mahller YY, Collins MH, Kim MO, Nobukuni T, Perentesis J, Cripe TP, Lane HA, Kozma SC, Thomas G, Ratner N: Effective in vivo targeting of the mammalian target of rapamycin pathway in malignant peripheral nerve sheath tumors. Mol Cancer Ther; 2008 May;7(5):1237-45
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  • [Title] Effective in vivo targeting of the mammalian target of rapamycin pathway in malignant peripheral nerve sheath tumors.
  • Malignant peripheral nerve sheath tumors (MPNST) are chemoresistant sarcomas with poor 5-year survival that arise in patients with neurofibromatosis type 1 (NF1) or sporadically.
  • We tested three drugs for single and combinatorial effects on collected MPNST cell lines and in MPNST xenografts.
  • The mammalian target of rapamycin complex 1 inhibitor RAD001 (Everolimus) decreased growth 19% to 60% after 4 days of treatment in NF1 and sporadic-derived MPNST cell lines.
  • Treatment of subcutaneous sporadic MPNST cell xenografts with RAD001 significantly, but transiently, delayed tumor growth, and decreased vessel permeability within xenografts.
  • RAD001 combined with the epidermal growth factor receptor tyrosine kinase inhibitor erlotinib caused additional inhibitory effects on growth and apoptosis in vitro, and a small but significant additional inhibitory effect on MPNST growth in vivo that were larger than the effects of RAD001 with doxorubicin.
  • The results support the consideration of RAD001 therapy in NF1 patient and sporadic MPNST.
  • The preclinical tests described allow rapid screening strata for drugs that block MPNST growth, prior to tests in more complex models, and should be useful to identify drugs that synergize with RAD001.

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  • (PMID = 18483311.001).
  • [ISSN] 1535-7163
  • [Journal-full-title] Molecular cancer therapeutics
  • [ISO-abbreviation] Mol. Cancer Ther.
  • [Language] ENG
  • [Grant] United States / NINDS NIH HHS / NS / NS028840-09; United States / NINDS NIH HHS / NS / R01 NS028840; United States / NINDS NIH HHS / NS / R01 NS028840-09; United States / NINDS NIH HHS / NS / R01 NS28840-17
  • [Publication-type] Comparative Study; Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Immunosuppressive Agents; 0 / Quinazolines; 9HW64Q8G6G / Everolimus; DA87705X9K / Erlotinib Hydrochloride; EC 2.7.- / Protein Kinases; EC 2.7.1.1 / MTOR protein, human; EC 2.7.1.1 / TOR Serine-Threonine Kinases; EC 2.7.1.1 / mTOR protein, mouse; EC 2.7.11.1 / Ribosomal Protein S6 Kinases, 70-kDa; EC 2.7.11.1 / ribosomal protein S6 kinase, 70kD, polypeptide 2; W36ZG6FT64 / Sirolimus
  • [Other-IDs] NLM/ NIHMS187346; NLM/ PMC2855168
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19. Grobmyer SR, Reith JD, Shahlaee A, Bush CH, Hochwald SN: Malignant Peripheral Nerve Sheath Tumor: molecular pathogenesis and current management considerations. J Surg Oncol; 2008 Mar 15;97(4):340-9
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  • [Title] Malignant Peripheral Nerve Sheath Tumor: molecular pathogenesis and current management considerations.
  • Malignant Peripheral Nerve Sheath Tumors (MPNSTs) are rare tumors that often occur in patients with neurofibromatosis 1.
  • Surgical resection represents the mainstay of treatment.
  • Radiation and chemotherapy have a role in selected patients with MPNST.
  • Accurate pathologic diagnosis remains a challenge in many cases of MPNST.
  • There are many recent advances in the understanding of the molecular pathogenesis of MPNST which represent the best opportunities to develop new strategies for management of patients with MPNST.
  • [MeSH-major] Nerve Sheath Neoplasms / genetics. Nerve Sheath Neoplasms / therapy
  • [MeSH-minor] Chemotherapy, Adjuvant. Chromosome Aberrations. Combined Modality Therapy. Humans. Neoplasm Recurrence, Local. Prognosis. Radiotherapy, Adjuvant

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  • [Copyright] (c) 2008 Wiley-Liss, Inc.
  • (PMID = 18286466.001).
  • [ISSN] 0022-4790
  • [Journal-full-title] Journal of surgical oncology
  • [ISO-abbreviation] J Surg Oncol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 100
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20. Jarmundowicz W, Jabłoński P, Załuski R: [Brachial plexus tumors--neurosurgical treatment]. Neurol Neurochir Pol; 2002 Sep-Oct;36(5):925-35
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Brachial plexus tumors--neurosurgical treatment].
  • Tumours of the brachial plexus according to present classification are included to soft tissue tumours.
  • Because of rare occurrence of these tumours diagnostic and operative experience is relatively small.
  • There is also a few number of publications regarding tumours of the brachial plexus.
  • Therefore the aim of the study was to present our experience in the surgical treatment of tumours of the brachial plexus basing on the material of 5 cases treated in the years 1997-2001.
  • In 3 cases tumours of the brachial plexus invaded the spinal canal through the intervertebral foramen and caused spinal cord compression (type A).
  • In 2 cases tumours involved only plexus elements (type B).
  • In 2 cases tumours were associated with neurofibromatosis type II.
  • Five cases of neural sheath tumours included 2 schwannomas, 2 neurofibromas and 1 schwannoma malignum.
  • In case of schwannomas and neurofibromas the surgical removal was radical without impairment of brachial plexus function.
  • In case of a giant schwannoma malignum tumor, which caused flaccid paresis and symptoms of insufficient blood, supply with severe pain in the upper limb radical extirpation was also possible.
  • In type A tumours in the first stage intraspinal part of the tumor was removed.
  • The result of treatment of benign tumours was very good with complete function recovery of the upper limb, pain disappearance and no symptoms of recurrence in the long postoperative period.
  • In case of malignant schwannoma in the early postoperative period both pain and symptoms of blood supply disturbances completely disappeared.
  • The patient died 12 months after the operation because of tumor dissemination.
  • Benign tumours of the brachial plexus can be effectively surgically treated using microsurgical techniques and, if necessary, nerve grafting.
  • In case of malignant tumours many authors also recommend surgery with optimal sparing of the brachial plexus function and subsequent radio and chemotherapy.
  • [MeSH-major] Brachial Plexus / surgery. Neurilemmoma / surgery. Neurofibroma / surgery. Peripheral Nervous System Neoplasms / surgery
  • [MeSH-minor] Adult. Female. Humans. Male. Middle Aged. Retrospective Studies. Time Factors. Treatment Outcome

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  • (PMID = 12523117.001).
  • [ISSN] 0028-3843
  • [Journal-full-title] Neurologia i neurochirurgia polska
  • [ISO-abbreviation] Neurol. Neurochir. Pol.
  • [Language] pol
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Poland
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21. Adamson DC, Cummings TJ, Friedman AH: Malignant peripheral nerve sheath tumor of the spine after radiation therapy for Hodgkin's lymphoma. Clin Neuropathol; 2004 Sep-Oct;23(5):245-55
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  • [Title] Malignant peripheral nerve sheath tumor of the spine after radiation therapy for Hodgkin's lymphoma.
  • We report the development of a malignant peripheral nerve sheath tumor (MPNST) in 2 patients after irradiation for Hodgkin's lymphoma.
  • Clinicians should be aware of this uncommon, but important fatal complication of radiation therapy.
  • He presented to our neurosurgery service with a left C6 radiculopathy 6 years later.
  • She did well with extensive radiotherapy until 5 years later when she developed severe right arm and chest pain secondary to recurrent lymphoma.
  • After aggressive radio- and chemotherapy, she presented to the neurosurgery service with a right Horner's syndrome, right C6 radiculopathy, and weakness of her right triceps and wrist extensors.
  • Both patients obtained magnetic resonance imaging revealing intradural extramedullary cervical nerve root associated mass lesions.
  • Two years after radiation therapy for his Hodgkin's lymphoma, the first patient underwent a C6 laminectomy at an outside institution for resection of a benign neurofibroma.
  • Four years later, he underwent a posterior C5-7 laminectomy with lateral mass plate fusion and partial excision of a recurrent mass diagnosed as a MPNST.
  • The second patient underwent a C5-6 hemilaminectomy and partial resection of a tumor also pathologically consistent with MPNST.
  • We present 2 case reports of patients who developed neurofibrosarcomatous tumors with malignant transformation after undergoing radiation therapy for Hodgkin's lymphoma.
  • Despite prompt surgical resection, these tumors exhibited aggressive behavior.
  • Numerous cases of soft tissue tumors have been described to arise in areas of prior radiation therapy; however, there have been rare reports of de novo MPNST after radiation therapy, especially in the setting of Hodgkin's lymphoma.
  • Postirradiation MPNST should be considered in the differential diagnosis of a painful, enlarging mass in a previously irradiated area.
  • [MeSH-major] Hodgkin Disease / radiotherapy. Lymphatic Irradiation / adverse effects. Neoplasms, Radiation-Induced / pathology. Nerve Sheath Neoplasms / etiology. Spinal Neoplasms / etiology
  • [MeSH-minor] Adult. Diagnosis, Differential. Fatal Outcome. Humans. Magnetic Resonance Imaging. Male


22. Amin MU, Shafique M: Isolated malignant peripheral nerve sheath tumor of retroperitoneum. J Coll Physicians Surg Pak; 2007 Apr;17(4):226-7
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  • [Title] Isolated malignant peripheral nerve sheath tumor of retroperitoneum.
  • A case of an isolated Malignant Peripheral Nerve Sheath Tumor (MPNST) of the retroperitoneum without neurofibromatosis is presented.
  • The tumor was located deep in the retroperitoneum with metastasis to the ribs.
  • Patient was further managed with radiotherapy and chemotherapy.
  • MPNST in such a location is very unusual.

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  • (PMID = 17462183.001).
  • [ISSN] 1022-386X
  • [Journal-full-title] Journal of the College of Physicians and Surgeons--Pakistan : JCPSP
  • [ISO-abbreviation] J Coll Physicians Surg Pak
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Pakistan
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23. Simon Z, Ress Z, Toldi J, Trauninger A, Miltényi Z, Illés A: Rare association of Hodgkin lymphoma, Graves' disease and myasthenia gravis complicated by post-radiation neurofibrosarcoma: coincidence or genetic susceptibility? Int J Hematol; 2009 May;89(4):523-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Rare association of Hodgkin lymphoma, Graves' disease and myasthenia gravis complicated by post-radiation neurofibrosarcoma: coincidence or genetic susceptibility?
  • With Hodgkin lymphoma (HL), other (autoimmune) diseases may occasionally occur or associate, whereas as a late treatment-complication, second tumour may develop.
  • Due to the residual mediastinal tumour CRu was declared but later on no progression/relapse could be proved by PET.
  • In 2000 Graves's disease, in 2001 myasthenia gravis was diagnosed, which showed resistance for immunosuppressant drugs, thus plasmapheresis, intravenous immunoglobulin treatments were applied.
  • In 2005, the residual mediastinal tumour started progressive growth, which leads to thoracotomy in which the tumour was removed, it was malignant peripheral nerve sheath tumour.
  • The disease showed progression despite the chemotherapy applied and the patient died in 2007 due to respiratory failure.
  • The etiologic role of genetic predisposition and immune regulatory disorder must definitely be thought of, as the possibility of mere coincidence is extremely small.
  • Malignant peripheral nerve sheath tumour is a rare complication of irradiation, which underlines the importance of the risk or/and response adapted therapy of HL.
  • [MeSH-major] Genetic Predisposition to Disease / genetics. Graves Disease / complications. Hodgkin Disease / complications. Myasthenia Gravis / complications. Neurofibrosarcoma / radiotherapy
  • [MeSH-minor] Adult. Autopsy. Fatal Outcome. Female. Humans. Positron-Emission Tomography. Tomography, X-Ray Computed


24. Zhang PJ, Brooks JS, Goldblum JR, Yoder B, Seethala R, Pawel B, Gorman JH, Gorman RC, Huang JH, Acker M, Narula N: Primary cardiac sarcomas: a clinicopathologic analysis of a series with follow-up information in 17 patients and emphasis on long-term survival. Hum Pathol; 2008 Sep;39(9):1385-95
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  • [Title] Primary cardiac sarcomas: a clinicopathologic analysis of a series with follow-up information in 17 patients and emphasis on long-term survival.
  • Although cardiac sarcomas are rare in comparison to their soft tissue counterparts, they are the second most common type of primary cardiac neoplasm.
  • There were 6 angiosarcomas, 6 myxofibrosarcomas, 3 malignant peripheral nerve sheath tumors, 3 leiomyosarcomas, 2 synovial sarcomas, 1 epithelioid hemangioendothelioma, 1 chondrosarcoma, 1 osteosarcoma, and 4 poorly differentiated sarcomas.
  • Six high-grade and 1 low-grade tumors were also treated with adjuvant chemotherapy and/or radiation.
  • In 17 patients with follow-up data, 6 of 12 patients with high-grade tumor died (4 within 5 days of the initial surgery, 1 in 21 months, and 1 in 131 months), and 1 patient with moderate-grade tumor and all 4 patients with low-grade tumor were alive without evidence of disease at the end of follow-up.
  • Tumor grade appeared to be prognostically important in cardiac sarcoma.
  • [MeSH-minor] Adolescent. Adult. Aged. Child. Child, Preschool. Combined Modality Therapy. Female. Follow-Up Studies. Humans. Infant. Male. Middle Aged. Survival Analysis

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  • (PMID = 18602663.001).
  • [ISSN] 1532-8392
  • [Journal-full-title] Human pathology
  • [ISO-abbreviation] Hum. Pathol.
  • [Language] eng
  • [Grant] United States / NHLBI NIH HHS / HL / R01 HL063954
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Other-IDs] NLM/ NIHMS522251; NLM/ PMC4081532
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25. Siveke JT, Sen Gupta R, Rieckhoff KU, Braumann D, Goldmann T: [Progressive paralysis caused by radiation-induced cervical malignant peripheral nerve sheath tumor]. HNO; 2003 Oct;51(10):825-8
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  • [Title] [Progressive paralysis caused by radiation-induced cervical malignant peripheral nerve sheath tumor].
  • HISTORY AND DIAGNOSIS: A 59-year old engineer was admitted to the hospital because of pain in his right collar region and the onset of incomplete paresis of the right arm.
  • Magnetic resonance tomography displayed an advanced tumour arising from the right paravertebral soft tissue.
  • Histological examination revealed a malignant peripheral nerve sheath tumor (MPNST).
  • Thirteen years before admission, the patient had a right-sided tumor-tonsillectomy of a squamous cell carcinoma and local radiation of a cystic squamous cell carcinoma in the ipsilateral cervical soft tissue.
  • CLINICAL COURSE AND THERAPY: In the following course, progressive neurological symptoms occurred including beginning paraplegia, right phrenic paralysis and a severe concomitant pain syndrome.
  • Due to the location and advanced tumor state, surgical treatment was not performed and palliative chemotherapy remained ineffective.
  • CONCLUSION: MPNST represents a rare entity which has been related to postoperative radiation.
  • Unusual neurological symptoms in anatomical regions of former radiation should therefore include neurogenic secondary malignancies in the differential diagnosis for early surgical intervention.
  • [MeSH-major] Carcinoma, Squamous Cell / radiotherapy. Head and Neck Neoplasms / diagnosis. Neoplasms, Multiple Primary / radiotherapy. Neoplasms, Radiation-Induced / diagnosis. Neoplasms, Second Primary / diagnosis. Nerve Sheath Neoplasms / diagnosis. Paraparesis / etiology. Radioisotope Teletherapy / adverse effects. Tonsillar Neoplasms / radiotherapy
  • [MeSH-minor] Arm / innervation. Combined Modality Therapy. Diagnosis, Differential. Humans. Magnetic Resonance Imaging. Male. Middle Aged. Radiotherapy, Adjuvant. Respiratory Paralysis / etiology. Tomography, X-Ray Computed


26. Shimizu J, Arano Y, Murata T, Ishikawa N, Yachi T, Nomura T, Minato H: A case of intrathoracic giant malignant peripheral nerve sheath tumor in neurofibromatosis type I (von Recklinghausen's disease). Ann Thorac Cardiovasc Surg; 2008 Feb;14(1):42-7
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  • [Title] A case of intrathoracic giant malignant peripheral nerve sheath tumor in neurofibromatosis type I (von Recklinghausen's disease).
  • She underwent surgery to alleviate respiratory and circulatory disorders caused by compression of the right lung and inferior vena cava due to the giant tumor.
  • Intraoperatively, the tumor was found to have originated from the 5th intercostal nerve.
  • The resected tumor was 20x17x15 cm in size and 2,300 g in weight.
  • It was histologically diagnosed as a malignant peripheral nerve sheath tumor.
  • Seven months after surgery, however, a recurrent tumor was found in the right thoracic cavity.
  • She died of rapid growth of recurrent tumor 3 months thereafter.
  • This tumor often complicates neurofibromatosis I and has a high frequency of local recurrence and distant metastasis, resulting in poor prognosis.
  • Neither an optimal extent of resection needed for complete resection of this tumor nor an optimal regimen of chemotherapy, radiotherapy, or other therapy for the tumor has yet been established.
  • [MeSH-major] Nerve Sheath Neoplasms / surgery. Neurofibromatosis 1 / complications. Thoracic Neoplasms / surgery
  • [MeSH-minor] Adult. Diagnosis, Differential. Fatal Outcome. Humans. Magnetic Resonance Imaging. Neoplasm Recurrence, Local. Tomography, X-Ray Computed


27. Malagón HD, Valdez AM, Moran CA, Suster S: Germ cell tumors with sarcomatous components: a clinicopathologic and immunohistochemical study of 46 cases. Am J Surg Pathol; 2007 Sep;31(9):1356-62
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  • [Title] Germ cell tumors with sarcomatous components: a clinicopathologic and immunohistochemical study of 46 cases.
  • The clinicopathologic features of 46 patients with germ cell tumors with sarcomatous components (GCTSC) involving either the primary site or their metastases were studied.
  • Twenty-three tumors arose in the mediastinum, 2 in the retroperitoneum, and 21 in the gonads.
  • The germ cell component consisted of pure mature or immature teratoma (23 cases), teratoma mixed with other seminomatous or nonseminomatous components (17), pure seminoma (2), intratubular germ cell neoplasia (1), and yolk sac tumor (1).
  • The SC included embryonal rhabdomyosarcoma (29), angiosarcoma (6), leiomyosarcoma (4), undifferentiated sarcoma (3), myxoid liposarcoma (1), malignant peripheral nerve sheath tumor (1), malignant "triton" tumor (1), and epithelioid hemangioendothelioma (1).
  • All patients were treated by cisplatinum-based chemotherapy plus other agents followed by surgery.
  • Thirty-two of 40 patients either died of tumor (25/40; 62.5%) or were alive with advanced, progressive disease (7/40; 17.5%), and only 8/40 (20%) were alive and free of disease between 5 to 40 months (mean=18 mo).
  • [MeSH-major] Immunohistochemistry. Mediastinal Neoplasms / diagnosis. Neoplasms, Germ Cell and Embryonal / diagnosis. Ovarian Neoplasms / diagnosis. Retroperitoneal Neoplasms / diagnosis. Sarcoma / diagnosis. Testicular Neoplasms / diagnosis
  • [MeSH-minor] Adolescent. Adult. Aged. Antineoplastic Combined Chemotherapy Protocols. Disease-Free Survival. Female. Follow-Up Studies. Humans. Male. Middle Aged. Neoplasm Metastasis. Neoplasm Staging. Orchiectomy. Ovariectomy. Time Factors. Treatment Outcome

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  • (PMID = 17721191.001).
  • [ISSN] 0147-5185
  • [Journal-full-title] The American journal of surgical pathology
  • [ISO-abbreviation] Am. J. Surg. Pathol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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28. Barkan B, Starinsky S, Friedman E, Stein R, Kloog Y: The Ras inhibitor farnesylthiosalicylic acid as a potential therapy for neurofibromatosis type 1. Clin Cancer Res; 2006 Sep 15;12(18):5533-42
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  • [Title] The Ras inhibitor farnesylthiosalicylic acid as a potential therapy for neurofibromatosis type 1.
  • We assessed the ability of FTS to reverse the transformed phenotype of neurofibromatosis type 1 (NF1)-associated tumor cell lines of malignant peripheral nerve sheath tumor (MPNST).
  • EXPERIMENTAL DESIGN: nf1 mutations were genotyped, allelic losses were analyzed, and neurofibromin expression levels were determined in MPNST cell lines ST88-14, S265P21, and 90-8.
  • The effects of FTS on GTP-bound Ras (Ras-GTP) and its prominent downstream targets, as well as on cell morphology, anchorage-dependent and anchorage-independent growth, and tumor growth in mice, were assessed.
  • RESULTS: The MPNST cell lines were biallelic, NF1 inactive, and neurofibromin deficient.
  • We show that FTS treatment shortened the relatively long duration of Ras activation and signaling to extracellular signal-regulated kinase, Akt, and RalA in all NF1-deficient MPNST cell lines (NF1 cells) to that observed in a non-NF1, normally expressing neurofibromin MPNST cell line.
  • NF1 tumor growth in a nude mouse model was inhibited by oral FTS.
  • CONCLUSIONS: FTS treatment of NF1 cells normalized Ras-GTP levels, resulting in reversal of the transformed phenotype and inhibition of tumor growth.
  • FTS may therefore be considered as a potential drug for the treatment of NF1.
  • [MeSH-major] Farnesol / analogs & derivatives. Neurofibromatosis 1 / drug therapy. Salicylates / therapeutic use. ras Proteins / antagonists & inhibitors
  • [MeSH-minor] Animals. Antineoplastic Agents / therapeutic use. Cell Adhesion / drug effects. Cell Proliferation / drug effects. Cytoskeleton / drug effects. Drug Evaluation. Genes, Neurofibromatosis 1 / physiology. Genes, ras / drug effects. Humans. Male. Mice. Mice, Nude. Mutation. Nerve Sheath Neoplasms / drug therapy. Nerve Sheath Neoplasms / genetics. Neurofibromin 1 / genetics. Signal Transduction / drug effects. Tumor Cells, Cultured. Xenograft Model Antitumor Assays

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  • (PMID = 17000690.001).
  • [ISSN] 1078-0432
  • [Journal-full-title] Clinical cancer research : an official journal of the American Association for Cancer Research
  • [ISO-abbreviation] Clin. Cancer Res.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Neurofibromin 1; 0 / Salicylates; 0 / farnesylthiosalicylic acid; 4602-84-0 / Farnesol; EC 3.6.5.2 / ras Proteins
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29. Zou CY, Smith KD, Zhu QS, Liu J, McCutcheon IE, Slopis JM, Meric-Bernstam F, Peng Z, Bornmann WG, Mills GB, Lazar AJ, Pollock RE, Lev D: Dual targeting of AKT and mammalian target of rapamycin: a potential therapeutic approach for malignant peripheral nerve sheath tumor. Mol Cancer Ther; 2009 May;8(5):1157-68
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  • [Title] Dual targeting of AKT and mammalian target of rapamycin: a potential therapeutic approach for malignant peripheral nerve sheath tumor.
  • The mammalian target of rapamycin (mTOR) pathway may constitute a potential target for the treatment of malignant peripheral nerve sheath tumors (MPNST).
  • Consequently, we hypothesized that dual phosphatidylinositol 3-kinase (PI3K)/AKT-mTOR blockade might be applicable for MPNST treatment.
  • Expression of activated mTOR downstream targets (p4EBP1 and pS6RP) and pAKT was evaluated immunohistochemically in a tissue microarray of human MPNSTs (n = 96) and benign neurofibromas (n = 31).
  • Results were analyzed by Wilcoxon rank-sum tests. mTOR and AKT pathways in human MPNST cell lines, and the effects of rapamycin (mTOR inhibitor), LY294002 (dual PI3K/mTOR inhibitor), and PI-103 (potent dual PI3K/AKT-mTOR inhibitor) on pathway activation were evaluated by Western blot.
  • Acridine orange staining/fluorescence-activated cell sorting analysis, electron microscopy, and Western blot evaluated autophagy induction. p4EBP1, pS6Rp, and pAKT levels were found to be significantly higher in MPNST versus neurofibroma (P < 0.05 for all markers).
  • mTOR and AKT pathways were found to be highly activated in MPNST cell lines.
  • MPNST cells were sensitive to rapamycin; however, rapamycin enhanced pAKT and peIF4E expression.
  • PI-103 abrogated MPNST cell growth and induced G(1) cell cycle arrest potentially through repression of cyclin D1.
  • PI-103 did not elicit apoptosis but significantly induced autophagy in MPNST cells.
  • These results suggest further study of combined PI3K/AKT and mTOR inhibition as a novel therapy for patients harboring MPNST.


30. Steins MB, Serve H, Zühlsdorf M, Senninger N, Semik M, Berdel WE: Carboplatin/etoposide induces remission of metastasised malignant peripheral nerve tumours (malignant schwannoma) refractory to first-line therapy. Oncol Rep; 2002 May-Jun;9(3):627-30
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  • [Title] Carboplatin/etoposide induces remission of metastasised malignant peripheral nerve tumours (malignant schwannoma) refractory to first-line therapy.
  • Malignant peripheral nerve tumours (MPNT), e.g. malignant schwannoma, represent a relatively rare tumour entity which is usually regarded as a member of the group of soft-tissue sarcomas and treated accordingly.
  • Both patients developed a partial remission with tumour reduction exceeding 50% after treatment with carboplatin in combination with etoposide (CE), 150 mg/m2 each, days 1-4 in 4-week intervals.
  • Complete resectability of lung metastases could be achieved, with histologic evidence for advanced tumour regression at the time of resection.
  • [MeSH-major] Carboplatin / therapeutic use. Drug Resistance, Neoplasm. Etoposide / therapeutic use. Neurilemmoma / drug therapy. Soft Tissue Neoplasms / drug therapy
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Antineoplastic Agents, Phytogenic / therapeutic use. Female. Humans. Male. Middle Aged. Neoplasm Metastasis. Time Factors. Tomography, X-Ray Computed. Treatment Outcome

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  • (PMID = 11956640.001).
  • [ISSN] 1021-335X
  • [Journal-full-title] Oncology reports
  • [ISO-abbreviation] Oncol. Rep.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Greece
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Antineoplastic Agents, Phytogenic; 6PLQ3CP4P3 / Etoposide; BG3F62OND5 / Carboplatin
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31. Galanis E, Okuno SH, Nascimento AG, Lewis BD, Lee RA, Oliveira AM, Sloan JA, Atherton P, Edmonson JH, Erlichman C, Randlev B, Wang Q, Freeman S, Rubin J: Phase I-II trial of ONYX-015 in combination with MAP chemotherapy in patients with advanced sarcomas. Gene Ther; 2005 Mar;12(5):437-45
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  • [Title] Phase I-II trial of ONYX-015 in combination with MAP chemotherapy in patients with advanced sarcomas.
  • We, therefore, undertook a phase I/II study of ONYX-015, days 1-5 every month administered intratumorally under radiographic guidance, in combination with MAP (mitomycin-C, doxorubicin, cisplatin) chemotherapy in patients with advanced sarcoma.
  • Sarcoma histologies were gastrointestinal stromal tumors (GIST, two patients), leiomyosarcoma (two patients), liposarcoma (one patient), and malignant peripheral nerve sheath tumor (1 patient).
  • Immunohistochemistry of the metastatic lesions prior to treatment showed that five out of six patients were positive for p53, while two patients also had mdm-2 overexpression.
  • ONYX-015 viral DNA was detected by quantitative PCR in the plasma of 5/6 patients on day 5 of the first cycle, and up to day 12 (7 days after the last viral dose) in one patient who had extended sampling for viral kinetics performed, suggesting viral replication in sarcoma tissue.
  • One patient with p53 mutation and MDM-2 amplification achieved a partial response to treatment that lasted 11 months.
  • In conclusion, intratumoral administration of ONYX-015 in combination with MAP chemotherapy is well tolerated with no significant toxicity due to ONYX-015 being encountered.
  • Detection of viral DNA in post treatment tumor specimens by ISH and detection of the ONYX-015 genome in the peripheral blood by quantitative PCR, up to 7 days after the last viral dose provide evidence for adenoviral replication.
  • [MeSH-major] Adenoviridae. Antineoplastic Agents / administration & dosage. Genetic Therapy / methods. Sarcoma / therapy
  • [MeSH-minor] Adult. Aged. Antibodies, Viral / blood. Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Cisplatin / administration & dosage. Combined Modality Therapy. DNA, Viral / analysis. DNA, Viral / blood. Doxorubicin / administration & dosage. Female. Humans. In Situ Hybridization. Injections, Intralesional. Male. Middle Aged. Mitomycin / administration & dosage. Viral Vaccines. Virus Replication

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  • (PMID = 15647767.001).
  • [ISSN] 0969-7128
  • [Journal-full-title] Gene therapy
  • [ISO-abbreviation] Gene Ther.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA 84388; United States / NCI NIH HHS / CA / U01CA 69912
  • [Publication-type] Clinical Trial; Clinical Trial, Phase I; Clinical Trial, Phase II; Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Antibodies, Viral; 0 / Antineoplastic Agents; 0 / DNA, Viral; 0 / ONYX015; 0 / Viral Vaccines; 50SG953SK6 / Mitomycin; 80168379AG / Doxorubicin; Q20Q21Q62J / Cisplatin; MAP protocol
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32. Slomiany MG, Dai L, Bomar PA, Knackstedt TJ, Kranc DA, Tolliver L, Maria BL, Toole BP: Abrogating drug resistance in malignant peripheral nerve sheath tumors by disrupting hyaluronan-CD44 interactions with small hyaluronan oligosaccharides. Cancer Res; 2009 Jun 15;69(12):4992-8
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  • [Title] Abrogating drug resistance in malignant peripheral nerve sheath tumors by disrupting hyaluronan-CD44 interactions with small hyaluronan oligosaccharides.
  • Malignant peripheral nerve sheath tumors (MPNST) develop in approximately 10% of neurofibromatosis type-1 patients and are a major contributing factor to neurofibromatosis-1 patient mortality and morbidity.
  • MPNSTs are multidrug resistant, and thus long-term patient survival rates are poor after standard doxorubicin or multiagent chemotherapies.
  • We show that the hyaluronan receptor CD44 forms complexes with multidrug transporters, BCRP (ABCG2) and P-glycoprotein (ABCB1), in the plasma membrane of human MPNST cells.
  • Treatment of MPNST cells with the hyaluronan oligomers causes disassembly of CD44-transporter complexes and induces internalization of CD44, BCRP, and P-glycoprotein.
  • Consequently, the oligomers suppress drug transporter activity and increase sensitivity to doxorubicin treatment in culture.
  • In vivo, systemic administration of hyaluronan oligomers inhibits growth of MPNST xenografts.
  • Moreover, the oligomers and doxorubicin act synergistically in vivo, in that combined suboptimal doses induce tumor regression to a greater extent than the additive effects of each agent alone.
  • These findings indicate that constitutive hyaluronan-CD44 interactions contribute to drug transporter localization and function at the plasma membrane, and that attenuating hyaluronan-CD44 interactions sensitizes MPNSTs to doxorubicin in vitro and in vivo.
  • These results also show the potential efficacy of hyaluronan oligomers, which are nontoxic and nonimmunogenic, as an adjuvant for chemotherapy in MPNST patients.
  • [MeSH-major] Antigens, CD44 / metabolism. Drug Resistance, Neoplasm. Hyaluronic Acid / metabolism. Nerve Sheath Neoplasms / drug therapy. Oligosaccharides / therapeutic use
  • [MeSH-minor] Cell Line, Tumor. Doxorubicin / therapeutic use. Drug Synergism. Humans. Immunoprecipitation. Microscopy, Confocal

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  • (PMID = 19470767.001).
  • [ISSN] 1538-7445
  • [Journal-full-title] Cancer research
  • [ISO-abbreviation] Cancer Res.
  • [Language] eng
  • [Grant] United States / NCRR NIH HHS / RR / C06 RR015455; United States / NCI NIH HHS / CA / R01 CA073839; United States / NCI NIH HHS / CA / R01 CA073839; United States / NCI NIH HHS / CA / R01 CA082867; United States / NCI NIH HHS / CA / R01 CA082867
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antigens, CD44; 0 / Oligosaccharides; 80168379AG / Doxorubicin; 9004-61-9 / Hyaluronic Acid
  • [Other-IDs] NLM/ NIHMS463086; NLM/ PMC3655760
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33. Jarkowski A 3rd: Possible contribution of aprepitant to ifosfamide-induced neurotoxicity. Am J Health Syst Pharm; 2008 Dec 1;65(23):2229-31
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  • SUMMARY: A 24-year-old white man diagnosed with a malignant peripheral nerve sheath tumor initially in the late 1990s was admitted to the hospital for treatment of a recurrence of the tumor in the supra-clavicular region.
  • With the fifth cycle of therapy, the patient suffered severe nausea and vomiting that required his readmission to the hospital.
  • With the initiation of the sixth cycle of chemotherapy, aprepitant was added to the existing antiemetic regimen of ondansetron and dexamethasone.
  • Since his symptoms resolved by morning, it was determined that the patient did not require treatment with methylene blue.
  • With the initiation of the seventh cycle of chemotherapy, aprepitant was again added to the standard antiemetic regimen of a corticosteroid and serotonin receptor antagonist.
  • The patient tolerated chemotherapy well without any signs or symptoms of neurotoxicity and was discharged four days later.
  • CONCLUSION: A 24-year-old patient treated with ifosfamide, carboplatin, and etoposide for a malignant peripheral nerve sheath tumor developed ifosfamide-induced neurotoxicity after the addition of aprepitant to a standard antiemetic regimen consisting of ondansetron and dexamethasone.
  • [MeSH-major] Antiemetics / adverse effects. Antineoplastic Combined Chemotherapy Protocols / adverse effects. Ifosfamide / adverse effects. Morpholines / adverse effects. Nerve Sheath Neoplasms / secondary. Neurotoxicity Syndromes / etiology
  • [MeSH-minor] Adult. Carboplatin / administration & dosage. Dexamethasone / administration & dosage. Drug Therapy, Combination. Etoposide / administration & dosage. Humans. Male. Nausea / prevention & control

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  • (PMID = 19020190.001).
  • [ISSN] 1535-2900
  • [Journal-full-title] American journal of health-system pharmacy : AJHP : official journal of the American Society of Health-System Pharmacists
  • [ISO-abbreviation] Am J Health Syst Pharm
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antiemetics; 0 / Morpholines; 1NF15YR6UY / aprepitant; 6PLQ3CP4P3 / Etoposide; 7S5I7G3JQL / Dexamethasone; BG3F62OND5 / Carboplatin; UM20QQM95Y / Ifosfamide
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34. García-Alvarez García F, Gil-Albarova J, Castiella T, García-Alvarez Alvarez F: [A case of multiple malignant schwannoma as the only disease in a 40-year-old patient: is this a new type of neurofibromatosis?]. Neurologia; 2000 Feb;15(2):81-4
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  • [Title] [A case of multiple malignant schwannoma as the only disease in a 40-year-old patient: is this a new type of neurofibromatosis?].
  • [Transliterated title] A propósito de un caso de schwannoma maligno múltiple como única patología en un paciente de 40 años: un nuevo tipo de neurofibromatosis?
  • We present the case of a 40-year-old man that presented a fast growing tumour on the external side of the left elbow.
  • The tumour was extirpated by means of marginal exerectomy.
  • The microscopic study corresponded to epithelioid malignant schwannoma, and the patient received radiotherapy and adjuvant chemotherapy.
  • During the following years two local recidives and two new malignant schwannomas, one in the left sciatic common trunk and another paraspinal one, appeared and were extirpated.
  • Radiotherapy and chemotherapy were again administered.
  • In the last year, four new tumours have appeared: in the supraclavicular space, right posterior costofrenic, left costovertebral, and in the inferior abdominal wall, none of them has been extirpated or has hystologic diagnosis at the moment, however radiologic findings suggest malignant schwannomas.
  • In this moment there is no neurologic deficiency except for the secondary ones to surgical procedures, and no neurofibromatosis types I to VII signs have been observed.
  • So, the possibility of a new neurofibromatosis type is appointed.
  • [MeSH-major] Neurilemmoma / pathology. Neurofibromatosis 1 / diagnosis. Peripheral Nervous System Neoplasms / pathology
  • [MeSH-minor] Adult. Diagnosis, Differential. Humans. Magnetic Resonance Imaging. Male

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  • (PMID = 10769537.001).
  • [ISSN] 0213-4853
  • [Journal-full-title] Neurología (Barcelona, Spain)
  • [ISO-abbreviation] Neurologia
  • [Language] spa
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] SPAIN
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35. Matsumoto H, Naito K, Hiragino T, Bara Y: [Retroperitoneal malignant schwannoma: a case report]. Hinyokika Kiyo; 2002 May;48(5):307-9
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  • [Title] [Retroperitoneal malignant schwannoma: a case report].
  • A 56-year-old woman was admitted with a complaint of a palpable left abdominal mass.
  • The left kidney was compressed by the tumor which measured 18 x 10 cm in size on computed tomographic scanning.
  • We diagnosed a left renal tumor or retroperitoneal tumor.
  • We performed radical nephrectomy with complete tumor resection on August 28, 1997.
  • The tumor had two satellites and was adherent to the renal capsule.
  • A yellowish-white solid tumor was macroscopically encapsulated by fibrous tissue and had cystic lesions filled with bloody fluid, weighed 940 g and was 18 x 11 x 8 cm in size.
  • Histopathological diagnosis was malignant schwannoma.
  • For poor responsiveness to chemotherapy and radiation therapy in this type of tumor, we did not perform adjuvant therapy.
  • [MeSH-major] Neurilemmoma / surgery. Retroperitoneal Neoplasms / surgery

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  • (PMID = 12094716.001).
  • [ISSN] 0018-1994
  • [Journal-full-title] Hinyokika kiyo. Acta urologica Japonica
  • [ISO-abbreviation] Hinyokika Kiyo
  • [Language] jpn
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Japan
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36. Mahller YY, Sakthivel B, Baird WH, Aronow BJ, Hsu YH, Cripe TP, Mehrian-Shai R: Molecular analysis of human cancer cells infected by an oncolytic HSV-1 reveals multiple upregulated cellular genes and a role for SOCS1 in virus replication. Cancer Gene Ther; 2008 Nov;15(11):733-41
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  • Oncolytic herpes simplex viruses (oHSVs) are promising anticancer therapeutics.
  • Five human malignant peripheral nerve sheath tumor cell lines, with differing sensitivity to oHSV, were infected with G207 for 6 h.

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  • (PMID = 18551144.001).
  • [ISSN] 1476-5500
  • [Journal-full-title] Cancer gene therapy
  • [ISO-abbreviation] Cancer Gene Ther.
  • [Language] ENG
  • [Grant] United States / NCI NIH HHS / CA / R01 CA114004-03; United States / NCI NIH HHS / CA / R01-CA114004; United States / NCI NIH HHS / CA / R01 CA114004; United States / NCI NIH HHS / CA / CA114004-02; United States / NCI NIH HHS / CA / R01 CA114004-01A2; United States / NCI NIH HHS / CA / T32 CA117846; United States / NCI NIH HHS / CA / CA114004-03; United States / NCI NIH HHS / CA / CA114004-01A2; United States / NCI NIH HHS / CA / R01 CA114004-02
  • [Publication-type] Comparative Study; Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] England
  • [Chemical-registry-number] 0 / SOCS1 protein, human; 0 / Suppressor of Cytokine Signaling Proteins
  • [Other-IDs] NLM/ NIHMS188160; NLM/ PMC2852536
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37. Chen L, Mao Y, Chen H, Zhou LF: Diagnosis and management of intracranial malignant peripheral nerve sheath tumors. Neurosurgery; 2008 Apr;62(4):825-32; discussion 832
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  • [Title] Diagnosis and management of intracranial malignant peripheral nerve sheath tumors.
  • OBJECTIVE: Intracranial malignant peripheral nerve sheath tumors (MPNSTs) are rare and generally carry a poor prognosis.
  • We have analyzed our experience with MPNSTs and conducted a review of the literature in an attempt to identify a rational approach to the management of these tumors.
  • METHODS: Eight patients underwent surgical treatment for intracranial MPNSTs during a 10-year period from 1996 to 2005.
  • The general strategy was to perform complete resection whenever possible and to provide adjuvant radiotherapy for residual tumor.
  • Chemotherapy was not used in this group.
  • Total tumor resection was achieved in five patients.
  • At this time, two have been recurrence-free for 3.5 and 5 years, respectively, and the other three patients had a mean postoperative survival of 7 months.
  • There was one case of near total (>90%) and two cases of partial (<90%) tumor removal; the postoperative survival rate was 4, 4, and 2 months, respectively.
  • CONCLUSION: MPNSTs are fast-growing, invasive tumors with rather unsatisfactory outcomes.
  • Total surgical resection seems to be the most effective therapeutic method, and radiotherapy may play a role in local control.
  • [MeSH-major] Cranial Nerve Neoplasms / diagnosis. Cranial Nerve Neoplasms / surgery. Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / surgery
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Female. Humans. Male. Middle Aged. Treatment Outcome

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  • (PMID = 18496188.001).
  • [ISSN] 1524-4040
  • [Journal-full-title] Neurosurgery
  • [ISO-abbreviation] Neurosurgery
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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38. Gallo A, Suriano M, Simonelli M, Ralli G, de Vincentiis M: Recurrent malignant schwannoma of the parapharyngeal space in neurofibromatosis type 1. Ear Nose Throat J; 2003 Nov;82(11):862-5
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  • [Title] Recurrent malignant schwannoma of the parapharyngeal space in neurofibromatosis type 1.
  • Malignant schwannoma is an aggressive tumor that carries a poor prognosis despite wide excision, chemotherapy, and radiotherapy.
  • Malignant schwannoma of the parapharyngeal space is an uncommon finding; to our knowledge, only four cases have been described in the literature during the past 30 years, and only one of them involved a patient who had clinical evidence of neurofibromatosis type 1.
  • In this article, we describe a new case of malignant schwannoma of the parapharyngeal space in a patient who had clinical evidence of neurofibromatosis type 1.
  • Following resection of the tumor and a total parotidectomy, the diagnosis was made on the basis of histology and immunohistochemistry.
  • The patient underwent postoperative chemotherapy with carboplatin and UP16.
  • However, 5 months following surgery, the tumor recurred and metastasized.
  • The IVA2 regimen slowed tumor growth, but 13 months after the initiation of therapy, the patient died of neoplastic cachexia.
  • Although chemotherapy is generally ineffective in most cases of malignant schwannoma, we did experience some positive results with the IVA2 protocol.
  • Therefore, we recommend that this combination be considered as a first-line adjuvant therapy following surgery or as a first-line therapy for patients with inoperable tumors.
  • [MeSH-major] Neurilemmoma / complications. Neurilemmoma / pathology. Neurofibromatosis 1 / complications. Pharyngeal Neoplasms / complications. Pharyngeal Neoplasms / pathology
  • [MeSH-minor] Adult. Fatal Outcome. Humans. Magnetic Resonance Imaging. Male. Neoplasm Recurrence, Local


39. Gonzalez LF, Lekovic GP, Eschbacher J, Coons S, Spetzler RF: A true malignant schwannoma of the eighth cranial nerve: case report. Neurosurgery; 2007 Aug;61(2):E421-2; discussion E422
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  • [Title] A true malignant schwannoma of the eighth cranial nerve: case report.
  • OBJECTIVE: The clinical presentation, pathology, treatment, and outcome of a 43-year-old woman with a malignant peripheral nerve sheath tumor arising from a benign schwannoma of the eighth cranial nerve are presented.
  • CLINICAL PRESENTATION: Initially, the tumor was debulked.
  • After finding malignant areas within the benign tumor, it was considered to be a malignant transformation of a previously benign tumor.
  • INTERVENTION: Aggressive total resection was obtained during a second-stage procedure.
  • Postoperatively, the tumor bed was radiated for palliation.
  • CONCLUSION: Despite surgery, radiation, and chemotherapy, the patient died rapidly as a result of disseminated metastatic disease.
  • [MeSH-major] Cranial Nerve Neoplasms / pathology. Neuroma, Acoustic / secondary. Vestibulocochlear Nerve / pathology

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  • (PMID = 17762727.001).
  • [ISSN] 1524-4040
  • [Journal-full-title] Neurosurgery
  • [ISO-abbreviation] Neurosurgery
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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40. Yone K, Ijiri K, Hayashi K, Yokouchi M, Takenouchi T, Manago K, Nerome Y, Ijichi O, Ikarimoto N, Komiya S: Primary malignant peripheral nerve sheath tumor of the cauda equina in a child case report. Spinal Cord; 2004 Mar;42(3):199-203
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Primary malignant peripheral nerve sheath tumor of the cauda equina in a child case report.
  • STUDY DESIGN: A case report of primary malignant peripheral nerve sheath tumor (MPNST) of the cauda equina in a child is presented, and the literature is reviewed.
  • OBJECTIVE: To discuss the problems involved in the treatment of primary intradural MPNSTs.
  • MRI revealed an intradural tumor at L3-L5 level.
  • Following laminectomy of L3, L4 and L5, the tumor was removed en bloc.
  • Based on pathological and immunohistological findings, the tumor was diagnosed as an MPNST.
  • RESULTS: Although adjuvant chemotherapy was administered local recurrence and cerebral and spinal metastases of the tumor were found 6 months after the operation.
  • Following additional incomplete removal of the recurrent tumor, radiation therapy was administered.
  • Although recurrent and metastatic tumors disappeared or diminished in size by radiation, tumors increased in size thereafter, despite additional adjuvant chemotherapy.
  • CONCLUSIONS: Reported clinical outcomes for patients with primary intradural MPNST are very poor.
  • Although no gold standard for the treatment of tumors has been established yet, surgical removal of tumors combined with postoperative high-dose radiation may be recommended.
  • [MeSH-major] Cauda Equina / pathology. Nerve Sheath Neoplasms / pathology. Nerve Sheath Neoplasms / secondary. Peripheral Nervous System Neoplasms / pathology

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  • (PMID = 15001982.001).
  • [ISSN] 1362-4393
  • [Journal-full-title] Spinal cord
  • [ISO-abbreviation] Spinal Cord
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
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41. Etienne-Mastroianni B, Falchero L, Chalabreysse L, Loire R, Ranchère D, Souquet PJ, Cordier JF: Primary sarcomas of the lung: a clinicopathologic study of 12 cases. Lung Cancer; 2002 Dec;38(3):283-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • PURPOSE: To study patients with primary sarcomas of the lung diagnosed in our pathology department in order to define their clinical characteristics, treatment, and prognosis.
  • PATIENTS: The study group consisted of 12 patients, with a mean age of 53 years.
  • Imaging findings consisted of: eight single peripheral opacities, three single parahilar opacities, and one lobar actelectasis.
  • The histologic diagnoses confirmed in all cases by detailed immunohistochemical study were leiomyosarcoma (7), monophasic synovial sarcoma (2), one case each of malignant peripheral nerve sheath tumor (MPNST), epithelioid sarcoma, and malignant fibrous histiocytoma.
  • Four patients received chemotherapy and two patients had radiation therapy postoperatively.
  • Treatment and prognosis do not differ from other soft tissue sarcomas.
  • [MeSH-minor] Adolescent. Adult. Aged. Aged, 80 and over. Female. Humans. Male. Middle Aged. Neoplasm Staging. Retrospective Studies. Survival Rate. Treatment Outcome

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  • (PMID = 12445750.001).
  • [ISSN] 0169-5002
  • [Journal-full-title] Lung cancer (Amsterdam, Netherlands)
  • [ISO-abbreviation] Lung Cancer
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Ireland
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42. Rekhi B, Jambhekar NA, Puri A, Agrawal M, Chinoy RF: Clinicomorphologic features of a series of 10 cases of malignant triton tumors diagnosed over 10 years at a tertiary cancer hospital in Mumbai, India. Ann Diagn Pathol; 2008 Apr;12(2):90-7
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  • [Title] Clinicomorphologic features of a series of 10 cases of malignant triton tumors diagnosed over 10 years at a tertiary cancer hospital in Mumbai, India.
  • A rhabdomyoblastic differentiation in a malignant peripheral nerve sheath tumor is unusual and is termed as a malignant triton tumor.
  • Distinct rhabdomyoblastic cells were identified in the areas of malignant peripheral nerve sheath tumor.
  • Surgery with adequate margins constituted the treatment mainstay with adjuvant chemotherapy and/or radiotherapy in individual cases.
  • Malignant triton tumor is an uncommon tumor associated with an aggressive behavior.
  • Surgery with clear margins is the treatment mainstay.
  • [MeSH-major] Nerve Sheath Neoplasms / pathology. Rhabdomyosarcoma / pathology
  • [MeSH-minor] Adolescent. Adult. Biomarkers, Tumor / metabolism. Female. Humans. Immunohistochemistry. Male. Middle Aged. Neoplasm Recurrence, Local. S100 Proteins / analysis. Treatment Outcome

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  • (PMID = 18325468.001).
  • [ISSN] 1092-9134
  • [Journal-full-title] Annals of diagnostic pathology
  • [ISO-abbreviation] Ann Diagn Pathol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / S100 Proteins
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43. Khorgami Z, Nasiri S, Rezakhanlu F, Sodagari N: Malignant schwannoma of anterior abdominal wall: report of a case. J Clin Med Res; 2009 Oct;1(4):233-6
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  • [Title] Malignant schwannoma of anterior abdominal wall: report of a case.
  • Malignant schwannoma of the anterior abdominal wall nerves is extremely rare.
  • Malignant peripheral nerve sheath tumors (MPNST) represent approximately 10% of all soft tissue sarcomas and it is found in 4% of patients with neurofibromatosis 1.
  • We present a case of malignant schwannoma in a 28-year-old female patient with neurofibromatosis 1.
  • She presented with a painful mass in the right upper quadrant of her abdomen.
  • The tumor location was in the abdominal wall in explorative laparatomy and malignant schwannoma was diagnosed in pathologic assessment.
  • The tumor recurred in 3 months and computed tomography showed two masses in the right side of abdominopelvic cavity.
  • In spite of administering chemotherapy after second surgery,the tumor recurred and magnetic resonance imaging finding showed a huge heterogeneously enhancing mass with adhesion to the inner side of the abdominal wall.
  • Tumor location and rapid recurrence was unique in our patient.
  • KEYWORDS: Malignant peripheral nerve sheath tumor; Malignant schwannoma; Abdominal wall.

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  • (PMID = 22461875.001).
  • [ISSN] 1918-3003
  • [Journal-full-title] Journal of clinical medicine research
  • [ISO-abbreviation] J Clin Med Res
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Canada
  • [Other-IDs] NLM/ PMC3299187
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44. Amin A, Saifuddin A, Flanagan A, Patterson D, Lehovsky J: Radiotherapy-induced malignant peripheral nerve sheath tumor of the cauda equina. Spine (Phila Pa 1976); 2004 Nov 1;29(21):E506-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Radiotherapy-induced malignant peripheral nerve sheath tumor of the cauda equina.
  • OBJECTIVES: To report a rare case of radiation-induced malignant peripheral nerve sheath tumor of the cauda equina 10 years after treatment for testicular seminoma.
  • SUMMARY OF BACKGROUND DATA: Development of malignant peripheral nerve sheath tumor after irradiation is well recognized and often associated with a dismal prognosis.
  • There have been isolated reports of malignant peripheral nerve sheath tumor developing in sites of previous irradiation for testicular seminoma.
  • Ten years previously, he had undergone right radical inguinal orchidectomy and adjuvant para-aortic radiotherapy as treatment for Stage I testicular seminoma.
  • Magnetic resonance imaging demonstrated an inoperable intra- and extradural tumor leading to significant cauda equina compression.
  • CT-guided biopsy revealed a diagnosis of malignant peripheral nerve sheath tumor, most likely due to previous radiotherapy.
  • His clinical condition did not improve, and he underwent a course of palliative chemotherapy.
  • CONCLUSIONS: Postirradiation malignant peripheral nerve sheath tumors are rare and occur in a population at high risk of developing second malignancies.
  • The authors report the fourth case resulting from adjuvant radiotherapy for testicular seminoma, with the present report being the first report of extensive intradural tumor leading to cauda equina syndrome.
  • [MeSH-major] Cauda Equina. Neoplasms, Radiation-Induced / etiology. Nerve Sheath Neoplasms / etiology. Peripheral Nervous System Neoplasms / etiology. Radiotherapy, Adjuvant / adverse effects
  • [MeSH-minor] Accidental Falls. Adult. Antineoplastic Agents / therapeutic use. Cervical Intraepithelial Neoplasia. Humans. Male. Orchiectomy. Palliative Care. Polyradiculopathy / etiology. Postoperative Complications / etiology. Retrospective Studies. Sciatica / complications. Seminoma / radiotherapy. Seminoma / surgery. Testicular Neoplasms / radiotherapy. Testicular Neoplasms / surgery

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  • (PMID = 15507791.001).
  • [ISSN] 1528-1159
  • [Journal-full-title] Spine
  • [ISO-abbreviation] Spine
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents
  • [Number-of-references] 21
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45. Li H, Zhang X, Fishbein L, Kweh F, Campbell-Thompson M, Perrin GQ, Muir D, Wallace M: Analysis of steroid hormone effects on xenografted human NF1 tumor schwann cells. Cancer Biol Ther; 2010 Oct 15;10(8):758-64
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  • [Title] Analysis of steroid hormone effects on xenografted human NF1 tumor schwann cells.
  • The neurofibroma, a common feature of neurofibromatosis type 1 (NF1), is a benign peripheral nerve sheath tumor that contains predominantly Schwann cells (SC).
  • This study examined the effects of estrogen and progesterone on proliferation and apoptosis in a panel of NF1 tumor xenografts.
  • SC-enriched cultures derived from three human NF1 tumor types (dermal neurofibroma, plexiform neurofibroma, and malignant peripheral nerve sheath tumor (MPNST)) were xenografted in sciatic nerves of ovariectomized scid /Nf1-/+ mice.
  • At the same time, mice were implanted with time-release pellets for systemic delivery of progesterone, estrogen or placebo.
  • Estrogen was found to increase the growth of all three MPNST xenografts.
  • Progesterone was associated with increased growth in two of the three MPNSTs, yet decreased growth of the other.
  • These findings indicate that human NF1 Schwann cells derived from some tumors show increased proliferation or decreased apoptosis in response to particular steroid hormones in a mouse xenograft model.
  • This suggests that anti-estrogen or anti-progesterone therapies may be worth considering for specific NF1 neurofibromas and MPNSTs.

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  • (PMID = 20699653.001).
  • [ISSN] 1555-8576
  • [Journal-full-title] Cancer biology & therapy
  • [ISO-abbreviation] Cancer Biol. Ther.
  • [Language] ENG
  • [Grant] United States / NINDS NIH HHS / NS / F30 NS043951; United States / NINDS NIH HHS / NS / 1F30NS43951
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, Non-P.H.S.
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Drug Implants; 0 / Estrogens; 0 / Ki-67 Antigen; 4G7DS2Q64Y / Progesterone
  • [Other-IDs] NLM/ PMC3093914
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46. Longhi A, Errani C, Magagnoli G, Alberghini M, Gambarotti M, Mercuri M, Ferrari S: High grade malignant peripheral nerve sheath tumors: outcome of 62 patients with localized disease and review of the literature. J Chemother; 2010 Dec;22(6):413-8
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  • [Title] High grade malignant peripheral nerve sheath tumors: outcome of 62 patients with localized disease and review of the literature.
  • Malignant peripheral nerve sheath tumours (MPNST) are rare sarcomas with one of the poorest prognoses of all the soft tissue sarcomas.
  • Information about adjuvant treatment is scarce and not homogeneous for this diagnosis.
  • We analyzed retrospectively the outcome of patients with localized high grade MPNST admitted to our institute from 1969 to 2008.
  • Of 62 evaluable patients, 23 were females and 39 males, median age 39 years (17-71), 22/62 had neurofibromatosis type I.
  • A total of 22/62 are alive; 26 patients had surgery alone, 18 received radiation therapy, 12 received radiation therapy and chemotherapy, and 6 received only adjuvant chemotherapy.
  • A positive trend for adjuvant radiation, but not for chemotherapy was observed according to univariate analysis only for disease-free survival and overall survival.
  • New drugs employed successfully in advanced mpNSt should be employed also in the adjuvant setting.
  • [MeSH-major] Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / therapy. Sarcoma / diagnosis. Sarcoma / therapy
  • [MeSH-minor] Adolescent. Adult. Aged. Chemotherapy, Adjuvant. Disease-Free Survival. Female. Humans. Male. Middle Aged. Retrospective Studies. Survival Rate. Treatment Outcome. Young Adult

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  • (PMID = 21303750.001).
  • [ISSN] 1973-9478
  • [Journal-full-title] Journal of chemotherapy (Florence, Italy)
  • [ISO-abbreviation] J Chemother
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Italy
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47. Oya N, Aoki J, Shinozaki T, Watanabe H, Takagishi K, Endo K: Preliminary study of proton magnetic resonance spectroscopy in bone and soft tissue tumors: an unassigned signal at 2.0-2.1 ppm may be a possible indicator of malignant neuroectodermal tumor. Radiat Med; 2000 May-Jun;18(3):193-8
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  • [Title] Preliminary study of proton magnetic resonance spectroscopy in bone and soft tissue tumors: an unassigned signal at 2.0-2.1 ppm may be a possible indicator of malignant neuroectodermal tumor.
  • PURPOSE: To evaluate the utility of proton magnetic resonance spectroscopy in bone and soft tissue tumors, especially whether or not the N-acetyl aspartate signal (NAA) could be recognized in neurogenic tumors.
  • MATERIALS AND METHODS: Forty-nine proton magnetic resonance spectroscopy studies were performed in 60 bone and soft tissue tumors.
  • RESULTS: An unassigned signal at about 2.0-2.1 ppm was recognized in six of 47 lesions: clear cell sarcoma (2/2), Ewing sarcoma (1/1), malignant fibrous histiocytoma (1/3), malignant schwannoma (1/1), and mucoepidermoid carcinoma (1/1).
  • Neuroblastoma (1/1), primitive neuroectodermal tumor (1/1), and malignant melanoma (1/1) after chemotherapy or radiotherapy did not show this signal.
  • This signal was not detected in neurofibroma (9/9), schwannoma (6/6), pheochromocytoma (2/2), or other mesenchymal tumors of non-neuroectodermal origin.
  • CONCLUSIONS: The assigned signal at about 2.0-2.1 ppm was detected in a small percentage of bone and soft tissue tumors and could be suggestive of an untreated malignant tumor of neuroectodermal origin.
  • [MeSH-major] Aspartic Acid / analogs & derivatives. Bone Neoplasms / diagnosis. Magnetic Resonance Spectroscopy. Neuroectodermal Tumors / diagnosis. Soft Tissue Neoplasms / diagnosis
  • [MeSH-minor] Adolescent. Adult. Aged. Aged, 80 and over. Child. Child, Preschool. Diagnosis, Differential. Female. Humans. Infant. Male. Middle Aged

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  • (PMID = 10972550.001).
  • [ISSN] 0288-2043
  • [Journal-full-title] Radiation medicine
  • [ISO-abbreviation] Radiat Med
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] JAPAN
  • [Chemical-registry-number] 30KYC7MIAI / Aspartic Acid; 997-55-7 / N-acetylaspartate
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48. Dartnell J, Pilling J, Ferner R, Cane P, Lang-Lazdunski L: Malignant triton tumor of the brachial plexus invading the left thoracic inlet: a rare differential diagnosis of pancoast tumor. J Thorac Oncol; 2009 Jan;4(1):135-7
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  • [Title] Malignant triton tumor of the brachial plexus invading the left thoracic inlet: a rare differential diagnosis of pancoast tumor.
  • Malignant triton tumor is a divergent malignant peripheral nerve sheath tumor with rhabdomyoblastic differentiation.
  • We report a case of malignant triton tumor arising in the brachial plexus of a 28-year-old women with neurofibromatosis type 1.
  • Fluorodeoxyglucose-positron emission tomography-computed tomography before excision demonstrated a tumor with a maximum standard uptake value of 21 at 4 hours postinjection.
  • The patient underwent complete excision of the tumor through median sternotomy and left supraclavicular approach.
  • Adjuvant radiotherapy and chemotherapy were planned but the patient died of metastatic disease within 3 months of surgical resection.
  • [MeSH-major] Brachial Plexus / pathology. Neurilemmoma / diagnosis. Pancoast Syndrome / diagnosis. Peripheral Nervous System Neoplasms / diagnosis. Thoracic Neoplasms / diagnosis
  • [MeSH-minor] Adult. Diagnosis, Differential. Fatal Outcome. Female. Fluorodeoxyglucose F18. Humans. Magnetic Resonance Imaging. Neoplasm Invasiveness. Neurofibromatosis 1 / complications. Positron-Emission Tomography. Radiopharmaceuticals. Tomography, X-Ray Computed

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  • (PMID = 19096322.001).
  • [ISSN] 1556-1380
  • [Journal-full-title] Journal of thoracic oncology : official publication of the International Association for the Study of Lung Cancer
  • [ISO-abbreviation] J Thorac Oncol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Radiopharmaceuticals; 0Z5B2CJX4D / Fluorodeoxyglucose F18
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49. Ferraresi V, Ciccarese M, Cercato MC, Nuzzo C, Zeuli M, Di Filippo F, Giannarelli D, Cognetti F: Gemcitabine at fixed dose-rate in patients with advanced soft-tissue sarcomas: a mono-institutional phase II study. Cancer Chemother Pharmacol; 2008 Dec;63(1):149-55
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  • [Title] Gemcitabine at fixed dose-rate in patients with advanced soft-tissue sarcomas: a mono-institutional phase II study.
  • PURPOSE: To explore the clinical activity and toxicity of gemcitabine infused at the fixed dose of 10 mg/m(2)/min over 100 min in patients with soft tissue sarcomas (STSs).
  • PATIENTS AND METHODS: Fourteen patients with advanced locally unresectable and/or metastatic, pretreated STSs (seven leiomyosarcoma, three malignant schwannoma, one synovialsarcoma, one malignant fibrous histiocytoma, one endometrial stromal cell sarcoma, one undifferentiated) were treated with gemcitabine 10 mg/m(2)/min/week over 100 min given for 3 weeks out of 4.
  • The median number of previous medical treatments for advanced disease was 1 (range 1-2).
  • Treatment was well tolerated and the main causes of dose-reduction or omission/delay were hematological and liver toxicities.
  • One patient (7%; 95% confidence interval: 0.2-33.9%) with a metastatic uterine leiomyosarcoma obtained a partial response that lasted for 6.5 months.
  • Three patients (two leiomyosarcoma and one schwannoma) (21%) obtained a stabilization of disease.
  • The median time to progression was 3.1 months (range 1.0-9.5).
  • Nevertheless, an interesting tumor growth control rate was observed in specific histological variants (i.e., leiomyosarcoma), thus confirming data from recent controlled clinical trials.
  • [MeSH-major] Antimetabolites, Antineoplastic / therapeutic use. Deoxycytidine / analogs & derivatives. Sarcoma / drug therapy. Soft Tissue Neoplasms / drug therapy
  • [MeSH-minor] Adult. Aged. Combined Modality Therapy. Disease Progression. Dose-Response Relationship, Drug. Female. Gastrointestinal Diseases / chemically induced. Hematologic Diseases / chemically induced. Humans. Male. Middle Aged

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  • (PMID = 18351342.001).
  • [ISSN] 1432-0843
  • [Journal-full-title] Cancer chemotherapy and pharmacology
  • [ISO-abbreviation] Cancer Chemother. Pharmacol.
  • [Language] eng
  • [Publication-type] Clinical Trial, Phase II; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antimetabolites, Antineoplastic; 0W860991D6 / Deoxycytidine; B76N6SBZ8R / gemcitabine
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50. Mattingly RR, Kraniak JM, Dilworth JT, Mathieu P, Bealmear B, Nowak JE, Benjamins JA, Tainsky MA, Reiners JJ Jr: The mitogen-activated protein kinase/extracellular signal-regulated kinase kinase inhibitor PD184352 (CI-1040) selectively induces apoptosis in malignant schwannoma cell lines. J Pharmacol Exp Ther; 2006 Jan;316(1):456-65
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  • [Title] The mitogen-activated protein kinase/extracellular signal-regulated kinase kinase inhibitor PD184352 (CI-1040) selectively induces apoptosis in malignant schwannoma cell lines.
  • Type 1 neurofibromatosis (NF1) is a common autosomal dominant disorder that results in neuroectodermal tumors.
  • The NF1 tumor-suppressor gene encodes neurofibromin, which includes a GTPase-activating domain for Ras inactivation.
  • Affinity purification showed N-Ras to be the predominant activated isoform of Ras in two independent neurofibrosarcoma cell lines from NF1 patients (lines ST88-14 and NF90-8).
  • These NF1 cells also demonstrated increased constitutive activity of the extracellular signal-regulated kinases 1 and 2 (ERK1,2) mitogen-activated protein (MAP) kinases compared with a sporadic malignant schwannoma cell line that maintains neurofibromin expression (STS-26T).
  • Thus, MAP kinase kinase (MEK) inhibitors may be a rational approach to NF1 therapy.
  • Hence, although PD184352 effectively killed neurofibrosarcoma cells, its effects on normal Schwann cells may limit its usefulness in the clinic.
  • [MeSH-major] Apoptosis / drug effects. Benzamides / pharmacology. Enzyme Inhibitors / pharmacology. Mitogen-Activated Protein Kinase Kinases / antagonists & inhibitors. Neurilemmoma / pathology
  • [MeSH-minor] Blotting, Western. Butadienes / pharmacology. Cell Line, Tumor. Cell Proliferation / drug effects. Flavonoids / pharmacology. Flow Cytometry. Gene Expression Regulation, Neoplastic / drug effects. Genes, p53 / genetics. Genes, ras / genetics. Humans. Mitogen-Activated Protein Kinase 7 / metabolism. Neurofibromatosis 1 / pathology. Nitriles / pharmacology. Phosphorylation. Reverse Transcriptase Polymerase Chain Reaction. Transfection


51. Zambrana F, Vicente F, García-Manrique T, Pereira S, Sáinz De Zaitigui J, De La Cruz Merino L: Primary intracranial malignant peripheral nerve sheath tumour responding to chemotherapy. Clin Transl Oncol; 2010 Mar;12(3):231-3
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  • [Title] Primary intracranial malignant peripheral nerve sheath tumour responding to chemotherapy.
  • Malignant peripheral nerve sheath tumours (MPNST) are a rare variety of soft tissue sarcomas (STS) arising from major peripheral nerve branches and typically located in the lower extremity, chest wall or the retroperitoneum.
  • It is a biologically aggressive neoplasm for which the treatment of choice is surgery, but usually requires a multimodality approach, having been generally labelled as chemoresistant.
  • We present a case of MPNST located intracranially with a good response to chemotherapy.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / drug therapy. Brain Neoplasms / pathology. Nerve Sheath Neoplasms / drug therapy

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  • (PMID = 20231129.001).
  • [ISSN] 1699-3055
  • [Journal-full-title] Clinical & translational oncology : official publication of the Federation of Spanish Oncology Societies and of the National Cancer Institute of Mexico
  • [ISO-abbreviation] Clin Transl Oncol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Italy
  • [Chemical-registry-number] 80168379AG / Doxorubicin; UM20QQM95Y / Ifosfamide
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52. Stark AM, Buhl R, Hugo HH, Mehdorn HM: Malignant peripheral nerve sheath tumours--report of 8 cases and review of the literature. Acta Neurochir (Wien); 2001;143(4):357-63; discussion 363-4
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  • [Title] Malignant peripheral nerve sheath tumours--report of 8 cases and review of the literature.
  • BACKGROUND: Though Malignant peripheral nerve sheath tumours (MPNST) are a rare entity accounting for 5-10% of soft-tissue sarcomas they are an important differential diagnosis to benign tumours of the peripheral nervous system regarding treatment and prognosis.
  • METHOD: We present our experience with eight patients who underwent surgery for MPNST at the Department of Neurosurgery between 10/1990 and 9/1999.
  • Two patients suffered from Neurofibromatosis type 1.
  • Paraffin embedded tumour specimens were immunohistochemically stained for S-100, p53 and Ki67/MIB-1.
  • Four tumours were localised at the head & neck region, three were found in the extremities and one tumour was located on the trunk.
  • All of these developed local recurrence with a mean disease free survival time of 10.6 months.
  • During follow up, three patients developed distant metastases located in the lung, liver and subcutaneous tissue.
  • Five out of eight patients died during follow-up with a mean survival time of 11.6 months after diagnosis.
  • The Ki67/MIB-1 proliferation index was detectable in all tumour samples, it differed from 10-30%.
  • INTERPRETATION: MPNST is a rare and fatal diagnosis in neurosurgery with high risk of local recurrence and occurence of distant metastases.
  • Though mulitimodal therapy including surgical resection and adjuvant radiotherapy including brachytherapy is available, the prognosis remains dismal.
  • Modern clinical studies and the development of effective chemotherapy is needed in order to gain control of the disease.
  • [MeSH-major] Head and Neck Neoplasms / mortality. Head and Neck Neoplasms / surgery. Neoplasm Recurrence, Local / mortality. Nerve Sheath Neoplasms / mortality. Nerve Sheath Neoplasms / surgery. Spinal Cord Neoplasms / mortality. Spinal Cord Neoplasms / surgery. Thoracic Neoplasms / mortality. Thoracic Neoplasms / surgery

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  • (PMID = 11437289.001).
  • [ISSN] 0001-6268
  • [Journal-full-title] Acta neurochirurgica
  • [ISO-abbreviation] Acta Neurochir (Wien)
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Austria
  • [Number-of-references] 5
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53. Terenziani M, D'Angelo P, Bisogno G, Boldrini R, Cecchetto G, Collini P, Conte M, De Laurentis T, Ilari I, Indolfi P, Inserra A, Piva L, Siracusa F, Spreafico F, Tamaro P, Lo Curto M: Teratoma with a malignant somatic component in pediatric patients: the Associazione Italiana Ematologia Oncologia Pediatrica (AIEOP) experience. Pediatr Blood Cancer; 2010 Apr;54(4):532-7
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  • [Title] Teratoma with a malignant somatic component in pediatric patients: the Associazione Italiana Ematologia Oncologia Pediatrica (AIEOP) experience.
  • BACKGROUND: Teratoma with a malignant somatic component (TMSC) is rare but described in adults, whereas information on pediatric presentation is sparse.
  • PROCEDURE: The Associazione Italiana Ematologia Oncologia Pediatrica identified 14 cases of TMSC.
  • RESULTS: The series (9 female, 5 male) showed the following disease: testis (2), sacrococcygeal (3), ovary (3), retroperitoneum (3), mediastinum (2), and foot soft tissue (1).
  • Distribution of the somatic component was: carcinoma (4), pancreatic neuroendocrine tumor (1), neuroblastoma (3), rhabdomyosarcoma (3), rhabdomyosarcoma plus liposarcoma, chondrosarcoma, neurogenic sarcoma (1), chondrosarcoma plus neuroectodermal sarcoma (1), malignant peripheral nerve sheath tumor (1).
  • CONCLUSIONS: Prognosis for germ cell tumors (GCTs) containing MSC is worse than that for GCTs.
  • The pediatric disease appears to be more heterogeneous in tumor site distribution and MSC histology than in adults.
  • Chemotherapy optimized for histology should include reagents directed to the somatic malignancy, if chemosensitive.
  • Malignant GCT warrants GCT-directed chemotherapy.
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Female. Humans. Infant. Infant, Newborn. Italy. Male. Neoplasm Staging. Prognosis. Retrospective Studies. Treatment Outcome


54. Kasper B, Lehnert T, Bernd L, Mechtersheimer G, Goldschmidt H, Ho AD, Egerer G: High-dose chemotherapy with autologous peripheral blood stem cell transplantation for bone and soft-tissue sarcomas. Bone Marrow Transplant; 2004 Jul;34(1):37-41
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  • [Title] High-dose chemotherapy with autologous peripheral blood stem cell transplantation for bone and soft-tissue sarcomas.
  • The role of high-dose chemotherapy (HDCT) with PBSCT in the treatment of bone and soft-tissue sarcomas is not established.
  • In total, 27 patients (15 female, median age at TPL 30.6 years (range: 13-59)) were analyzed (Ewing sarcoma family n=8, osteosarcoma n=6, MPNST (malignant peripheral nerve sheath tumor) n=4, synovial sarcoma n=3, liposarcoma n=2, leiomyosarcoma n=2, rhabdomyosarcoma n=1, meningosarcoma n=1).
  • Following chemotherapy and surgery complete remission (CR) (n=9), partial remission (PR) (n=10), stable disease (SD) (n=2) and progressive disease (PD) (n=6) were reached prior HDCT.
  • Although the role of HDCT in the treatment of sarcomas is not defined, a subgroup of patients who achieved CR before HDCT could benefit from this therapy.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Bone Neoplasms / therapy. Peripheral Blood Stem Cell Transplantation / methods. Sarcoma / therapy
  • [MeSH-minor] Adolescent. Adult. Female. Humans. Male. Middle Aged. Remission Induction. Retrospective Studies. Survival Analysis. Transplantation, Autologous. Treatment Outcome


55. Capote A, Escorial V, Reina T, Muñoz-Guerra MF, Nieto S, Naval L: Primary malignant schwannoma of the cervical plexus with melanocytic differentiation. Int J Oral Maxillofac Surg; 2006 Aug;35(8):767-71
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  • [Title] Primary malignant schwannoma of the cervical plexus with melanocytic differentiation.
  • Primary malignant schwannomas are rare neoplasms of nerve sheath origin, especially in the location of the head and neck where few cases are described in the literature.
  • These tumours may pose a diagnostic dilemma in the work-up of a neck mass.
  • The case presented here is of a malignant schwannoma that originated in the cervical plexus with the rare histological feature of melanocytic differentiation.
  • Histopathological examination with immunostaining techniques is essential for the diagnosis of these tumours.
  • The management of these neoplasms is still controversial, although the treatment of choice is radical surgical excision of the lesion.
  • The role of postoperative radiotherapy or chemotherapy is not clear, although some authors recommend its use to prevent local recurrence, for unresectable recurrent tumours or in cases of distant metastases.
  • [MeSH-major] Cervical Plexus / pathology. Head and Neck Neoplasms / pathology. Neurilemmoma / pathology
  • [MeSH-minor] Aged. Humans. Male. Neck Dissection / methods. Survival Rate. Treatment Outcome

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  • (PMID = 16580816.001).
  • [ISSN] 0901-5027
  • [Journal-full-title] International journal of oral and maxillofacial surgery
  • [ISO-abbreviation] Int J Oral Maxillofac Surg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Denmark
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56. Ambrosini G, Sambol EB, Carvajal D, Vassilev LT, Singer S, Schwartz GK: Mouse double minute antagonist Nutlin-3a enhances chemotherapy-induced apoptosis in cancer cells with mutant p53 by activating E2F1. Oncogene; 2007 May 24;26(24):3473-81
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  • [Title] Mouse double minute antagonist Nutlin-3a enhances chemotherapy-induced apoptosis in cancer cells with mutant p53 by activating E2F1.
  • MDM2 is a critical negative regulator of the p53 tumor suppressor protein.
  • Recently, small-molecule antagonists of MDM2, the Nutlins, have been developed to inhibit the p53-MDM2 interaction and activate p53 signaling.
  • However, half of human cancers have mutated p53 and they are resistant to Nutlin treatment.
  • Here, we report that treatment of the p53-mutant malignant peripheral nerve sheath (MPNST) and p53-null HCT116 cells with cisplatin (Cis) and Nutlin-3a induced a degree of apoptosis that was significantly greater than either drug alone.
  • Nutlin-3a also increased the cytotoxicity of both carboplatin and doxorubicin in a series of p53-mutant human tumor cell lines.
  • In the human dedifferentiated liposarcoma cell line (LS141) and the p53 wild-type HCT116 cells, Nutlin-3a induced downregulation of E2F1 and this effect appeared to be proteasome dependent.
  • In contrast, in MPNST and HCTp53-/- cells, Nutlin-3a inhibited the binding of E2F1 to MDM2 and induced transcriptional activation of free E2F1 in the presence of Cis-induced DNA damage.
  • Downregulation of E2F1 by small interfering RNA significantly decreased the level of apoptosis induced by Cis and Nutlin-3a treatment.
  • Moreover, expression of a dominant-negative form of E2F1 rescued cells from apoptosis, whereas cells overexpressing wild-type E2F1 showed an increase in cell death.
  • Nutlin-3a therefore may provide a therapeutic benefit in tumors with mutant p53 provided it is combined with chemotherapy.
  • [MeSH-major] Antineoplastic Agents / pharmacology. Apoptosis / drug effects. E2F1 Transcription Factor / metabolism. Imidazoles / pharmacology. Piperazines / pharmacology. Tumor Suppressor Protein p53 / genetics
  • [MeSH-minor] Animals. Carboplatin / pharmacology. Cisplatin / pharmacology. DNA-Binding Proteins / drug effects. DNA-Binding Proteins / metabolism. Doxorubicin / pharmacology. Humans. Mice. Nuclear Proteins / drug effects. Nuclear Proteins / metabolism. Proto-Oncogene Proteins c-bcl-2 / drug effects. Proto-Oncogene Proteins c-bcl-2 / metabolism. Proto-Oncogene Proteins c-mdm2 / metabolism. Transcription, Genetic. Tumor Cells, Cultured. Tumor Suppressor Proteins / drug effects. Tumor Suppressor Proteins / metabolism

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  • (PMID = 17146434.001).
  • [ISSN] 0950-9232
  • [Journal-full-title] Oncogene
  • [ISO-abbreviation] Oncogene
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / DNA-Binding Proteins; 0 / E2F1 Transcription Factor; 0 / E2f1 protein, mouse; 0 / Imidazoles; 0 / Nuclear Proteins; 0 / PMAIP1 protein, human; 0 / Piperazines; 0 / Proto-Oncogene Proteins c-bcl-2; 0 / Tumor Suppressor Protein p53; 0 / Tumor Suppressor Proteins; 0 / nutlin 3; 0 / tumor suppressor protein p73; 80168379AG / Doxorubicin; BG3F62OND5 / Carboplatin; EC 6.3.2.19 / MDM2 protein, human; EC 6.3.2.19 / Proto-Oncogene Proteins c-mdm2; Q20Q21Q62J / Cisplatin
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57. Mandić A, Djurdjević S, Popov M, Krnojelać D, Kukić B: Retroperitoneal malignant schwannoma and peritoneal malignant mesothelioma: a case report. J BUON; 2004 Jan-Mar;9(1):91-4
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  • [Title] Retroperitoneal malignant schwannoma and peritoneal malignant mesothelioma: a case report.
  • Malignant schwannoma and peritoneal malignant mesothelioma (MM) are very rare tumors.
  • Schwannoma or neurilemmoma-benign or malignant-do not arise from the nerves, but from the supporting Schwann cells.
  • Malignant peripheral nerve sheath tumors (MPNSTs) commonly are large in size.
  • A malignant schwannoma was diagnosed in a 52-yearold woman, which was surgically treated.
  • The patient was operated on for second time and MM was diagnosed as second primary tumor, along with recurrence of the malignant schwannoma.
  • The patient received postoperative adjuvant external beam radiotherapy and chemotherapy.
  • Despite combined-modality treatment the disease progressed and the patient was operated on for third time 2.5 years after the first operation with partial tumor resection.
  • She died 3 years after the first diagnosis.
  • Early diagnosis of these two types of tumors is very difficult because of unspecific clinical symptoms.
  • Singlemodality therapy of these tumors has shown poor results.

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  • (PMID = 17385835.001).
  • [ISSN] 1107-0625
  • [Journal-full-title] Journal of B.U.ON. : official journal of the Balkan Union of Oncology
  • [ISO-abbreviation] J BUON
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Greece
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58. Yoo NJ, Kim HS, Kim SY, Park WS, Park CH, Jeon H, Jung ES, Lee JY, Lee SH: Immunohistochemical analysis of Smac/DIABLO expression in human carcinomas and sarcomas. APMIS; 2003 Mar;111(3):382-8
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  • Alteration of apoptosis is essential for cancer development, and cancer cell death by radiation and chemotherapy is largely dependent upon apoptosis.
  • In this study, archival tissues of 100 carcinomas and 50 sarcomas from various origins were analyzed by immunohistochemistry for the expression of Smac/DIABLO.
  • Smac/DIABLO is expressed in 11 of 50 (22%) sarcomas, including 2 of 8 malignant schwannomas, 5 of 11 rhabdomyosarcomas, 2 of 7 malignant fibrous histiocytomas, 1 of 6 leiomyosarcomas, 0 of 8 angiosarcomas, 0 of 8 liposarcomas, and 1 of 2 Ewing's sarcomas.
  • These data demonstrated that Smac/DIABLO expression levels vary depending on the individual cancer types.

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  • (PMID = 12752217.001).
  • [ISSN] 0903-4641
  • [Journal-full-title] APMIS : acta pathologica, microbiologica, et immunologica Scandinavica
  • [ISO-abbreviation] APMIS
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Denmark
  • [Chemical-registry-number] 0 / Carrier Proteins; 0 / DIABLO protein, human; 0 / Intracellular Signaling Peptides and Proteins; 0 / Mitochondrial Proteins
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59. Fleshman R, Mayerson J, Wakely PE Jr: Fine-needle aspiration biopsy of high-grade sarcoma: a report of 107 cases. Cancer; 2007 Dec 25;111(6):491-8
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  • They also searched their tissue database for all HGS cases that had prior FNA biopsy findings.
  • RESULTS: A total of 107 FNA samples from 98 patients (age range, 13-90 years, with a male:female ratio of 1:1) had an FNA diagnosis of HGS, or had HGS and a prior FNA diagnosis of another entity.
  • The positive predictive value of an FNA diagnosis of HGS was 97% (88 of 91 cases).
  • Fifty-four cases were diagnosed as HGS, not otherwise specified, 8 as myxofibrosarcoma, 8 as osteosarcoma, 5 as malignant peripheral nerve sheath tumor, 5 as leiomyosarcoma, 4 as Ewing sarcoma, 4 as liposarcoma, 2 as epithelioid sarcoma, and 1 as angiosarcoma.
  • Approximately 71% of patients presented with a primary tumor, 23% with disease recurrence, and 7% with metastasis.
  • FNA diagnosis was confirmed histologically in 88% of cases, clinically in 7% of cases, and cytogenetically in 1% of cases; 3% of cases had false-positive results and 1 patient was lost to follow-up.
  • Sixteen of 19 patients received neoadjuvant chemotherapy based on the FNA diagnosis alone.
  • CONCLUSIONS: A cytopathologic diagnosis of HGS was found to be accurate in 88 of 97 cases (91%) with follow-up.
  • A FNA biopsy diagnosis of HGS appears to be clinically reliable in a high percentage of cases when used in close conjunction with the orthopedic team.
  • [MeSH-major] Biopsy, Fine-Needle. Cytodiagnosis. Sarcoma / diagnosis. Soft Tissue Neoplasms / diagnosis
  • [MeSH-minor] Adolescent. Adult. Aged. Aged, 80 and over. Antineoplastic Agents / therapeutic use. Bone Neoplasms / diagnosis. Female. Humans. Male. Middle Aged. Neoadjuvant Therapy. Predictive Value of Tests. Reproducibility of Results

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  • (PMID = 17941014.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Evaluation Studies; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents
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60. Lee CS, Huh JS, Chang JW, Park JK: The early detection of recurrence of malignant peripheral nerve sheath tumor by frequent magnetic resonance imaging. J Korean Neurosurg Soc; 2010 Jan;47(1):51-4
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  • [Title] The early detection of recurrence of malignant peripheral nerve sheath tumor by frequent magnetic resonance imaging.
  • Surgery has a key role in the treatment of malignant peripheral nerve sheath tumors (MPNSTs), but the resectability of paraspinal MPNSTs is only 20%.
  • Therefore, spinal MPNSTs show frequent recurrence and poor prognosis.
  • Local recurrence is much more common than metastasis for MPNSTs, and surgery still has a key role in the treatment of local recurrence.
  • However, no evidence-based follow-up protocol has been established for MPNST.
  • The authors performed gross total resection in a 34-year-old woman presented with thoracic MPNST.
  • Adjuvant radiotherapy and chemotherapy were not administered since these adjuvant therapies generally do not improve survival in MPNST and may cause additional neurovascular damage.
  • The tumor recurred locally on two occasions without overt symptoms at 21 and 24 months postoperatively.

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  • [Cites] Neurosurg Focus. 2007;22(6):E6 [17613223.001]
  • [Cites] Neurosurg Focus. 2007;22(6):E13 [17613204.001]
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  • (PMID = 20157379.001).
  • [ISSN] 1598-7876
  • [Journal-full-title] Journal of Korean Neurosurgical Society
  • [ISO-abbreviation] J Korean Neurosurg Soc
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Korea (South)
  • [Other-IDs] NLM/ PMC2817516
  • [Keywords] NOTNLM ; Follow-up / Malignant peripheral nerve sheath tumor / Recurrence
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61. Manger T, Pross M, Haeckel C, Lippert H: Malignant peripheral nerve sheath tumor of the esophagus. Dig Surg; 2000;17(6):627-631
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  • [Title] Malignant peripheral nerve sheath tumor of the esophagus.
  • BACKGROUND: Sarcomas of the esophagus are rare representing 0.1-1.5% of all esophageal tumors.
  • We report a case of malignant peripheral nerve sheath tumor (MPNST) of the esophagus in a 60-year-old woman.
  • METHODS: The diagnosis was made preoperatively on endoscopic biopsy and confirmed after tumor resection by immunohistochemistry as well as electron microscopy.
  • RESULTS: Our therapeutic concept for the first case of a high-grade MPNST (malignant schwannoma) of the esophagus resulted in a recurrence-free interval of 4 years.
  • CONCLUSION: The therapy of choice was abdominal-thoracic en bloc esophagectomy with tumor-free resection margins and esophageal reconstruction with the stomach.
  • After R0-resection we found no indication for adjuvant radio- and/or chemotherapy.
  • [MeSH-major] Esophageal Neoplasms / surgery. Esophagectomy. Nerve Sheath Neoplasms / surgery

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  • [Copyright] Copyright 2000 S. Karger AG, Basel.
  • (PMID = 11155010.001).
  • [ISSN] 0253-4886
  • [Journal-full-title] Digestive surgery
  • [ISO-abbreviation] Dig Surg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Switzerland
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62. Perrin RG, Guha A: Malignant peripheral nerve sheath tumors. Neurosurg Clin N Am; 2004 Apr;15(2):203-16
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  • [Title] Malignant peripheral nerve sheath tumors.
  • The rarity of MPNSTs and the lack of any singular diagnostic radiologic or pathologic signature lead to several management challenges.
  • These tumors are best managed as part ofa multidisciplinary team so as to optimize patient care and facilitate research.
  • Suspicion of an MPNST based on clinical or radiologic alteration of a soft tissue mass in proximity to a peripheral nerve, especially in the context of NF I, should lead to referral to such a tertiary center.
  • Early diagnosis followed by oncologic surgery to obtain tumor-free margins provides the best chance for long-term cure.
  • Current adjuvant therapy with radiation and chemotherapy is suboptimal.
  • There have been major inroads toward the molecular biologic understanding of MPNSTs,with several biologic targets that are of potential therapeutic interest.
  • [MeSH-major] Neurofibromatosis 1 / pathology. Neurofibromatosis 1 / surgery. Peripheral Nervous System Neoplasms / pathology. Peripheral Nervous System Neoplasms / surgery

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  • (PMID = 15177319.001).
  • [ISSN] 1042-3680
  • [Journal-full-title] Neurosurgery clinics of North America
  • [ISO-abbreviation] Neurosurg. Clin. N. Am.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 125
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63. Moon SJ, Lee JK, Seo BR, Kim JH, Kim SH, Lee KH, Lee MC: An intraosseous malignant peripheral nerve sheath tumor of the cervical spine: a case report and review of the literature. Spine (Phila Pa 1976); 2008 Sep 1;33(19):E712-6
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  • [Title] An intraosseous malignant peripheral nerve sheath tumor of the cervical spine: a case report and review of the literature.
  • OBJECTIVES: To report a rare case of intraosseous malignant peripheral nerve sheath tumors (MPNST), and review the pertinent medical literature.
  • SUMMARY OF BACKGROUND DATA: The spinal MPNST that develops from spinal nerve roots and secondary bony erosion is well-known entity.
  • However, primary intraosseous MPNSTs of the spine are extremely rare.
  • METHODS: A 41-year-old male presented with a 1-month history of radiating pain to his right shoulder and arm.
  • Complete excision of the tumor and posterior stabilization were performed through a posterior approach.
  • The tumor was noted to originate from the posterior element of C7.
  • RESULTS: The histopathology was diagnostic for a MPNST.
  • Adjuvant chemotherapy was administered after surgery.
  • CONCLUSION: We report an intraosseous MPNST of the cervical spine.
  • Complete surgical excision and adjuvant chemotherapy resulted in a good functional outcome.
  • MPNST should be added to the differential diagnosis of primary bone tumors causing spinal cord compression.
  • [MeSH-major] Cervical Vertebrae / pathology. Nerve Sheath Neoplasms / pathology. Spinal Neoplasms / pathology
  • [MeSH-minor] Adult. Antigens, CD / metabolism. Antigens, CD34 / metabolism. Biomarkers, Tumor / metabolism. Cell Adhesion Molecules / metabolism. Chemotherapy, Adjuvant. Combined Modality Therapy. Disease-Free Survival. Humans. Ki-67 Antigen / metabolism. Magnetic Resonance Imaging. Male. S100 Proteins / metabolism. Spinal Cord Compression / etiology. Spinal Cord Compression / pathology. Vimentin / metabolism

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  • (PMID = 18758353.001).
  • [ISSN] 1528-1159
  • [Journal-full-title] Spine
  • [ISO-abbreviation] Spine
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antigens, CD; 0 / Antigens, CD34; 0 / Biomarkers, Tumor; 0 / CD99 protein, human; 0 / Cell Adhesion Molecules; 0 / Ki-67 Antigen; 0 / S100 Proteins; 0 / Vimentin
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64. Kinebuchi Y, Noguchi W, Igawa Y, Nishizawa O: Recurrent retroperitoneal malignant nerve sheath tumor associated with neurofibromatosis type 1 responding to carboplatin and etoposide combined chemotherapy. Int J Clin Oncol; 2005 Oct;10(5):353-6
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  • [Title] Recurrent retroperitoneal malignant nerve sheath tumor associated with neurofibromatosis type 1 responding to carboplatin and etoposide combined chemotherapy.
  • A 25-year-old man was referred to our hospital with left flank pain, and computed tomography (CT) and magnetic resonance imaging (MRI) revealed large retroperitoneal masses.
  • Physical examination revealed many café-au-lait spots and superficial neurofibromas, and a diagnosis of neurofibromatosis type 1 (von Recklinghausen's disease) was made.
  • The tumor was resected, and the pathological diagnosis was malignant peripheral nerve sheath tumor (MPNST).
  • He received four courses of chemotherapy with carboplatin and etoposide, and the metastatic lung lesions were markedly decreased.
  • After chemotherapy, complete resection of the remaining lung lesions was performed, and there has been no recurrence to date.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Nerve Sheath Neoplasms / drug therapy. Neurofibromatosis 1 / complications. Peripheral Nervous System Neoplasms / drug therapy. Retroperitoneal Neoplasms / drug therapy

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  • [Cites] Crit Rev Oncol Hematol. 1995 Oct;20(3):193-201 [8748009.001]
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  • (PMID = 16247664.001).
  • [ISSN] 1341-9625
  • [Journal-full-title] International journal of clinical oncology
  • [ISO-abbreviation] Int. J. Clin. Oncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 6PLQ3CP4P3 / Etoposide; BG3F62OND5 / Carboplatin
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65. Carli M, Ferrari A, Mattke A, Zanetti I, Casanova M, Bisogno G, Cecchetto G, Alaggio R, De Sio L, Koscielniak E, Sotti G, Treuner J: Pediatric malignant peripheral nerve sheath tumor: the Italian and German soft tissue sarcoma cooperative group. J Clin Oncol; 2005 Nov 20;23(33):8422-30
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  • [Title] Pediatric malignant peripheral nerve sheath tumor: the Italian and German soft tissue sarcoma cooperative group.
  • PURPOSE: To assess the value of chemotherapy and radiotherapy in children with malignant peripheral nerve sheath tumors (MPNSTs) and to identify risk factors associated with outcome.
  • Seventeen percent of patients had neurofibromatosis type 1 (NF1).
  • Chemotherapy was administered to 74% of patients; radiotherapy was administered to 38% of patients.
  • RESULTS: With a median follow-up of 7 years, 5-year overall survival (OS) and progression-free survival (PFS) were 51% and 37%, respectively.
  • Univariate analysis identified IRS groups, size, invasiveness, primary site, age, and presence of NF1 as prognostic factors; multivariate analysis identified absence of NF1, tumor invasiveness T1, IRS groups I to II and extremity of primary site as independent favorable factors for OS.
  • The overall response rate to primary chemotherapy, including minor responses, in group III patients was 45%.
  • CONCLUSION: MPNST is an aggressive tumor for which complete surgical resection is the mainstay of successful treatment.
  • Postoperative radiotherapy may have a role in improving local control in patients with minimal residual tumor.
  • The reported responses to primary chemotherapy suggest that it may be effective in patients with tumor considered unresectable at diagnosis.
  • [MeSH-major] Nerve Sheath Neoplasms / drug therapy. Nerve Sheath Neoplasms / radiotherapy
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Child. Child, Preschool. Combined Modality Therapy. Disease-Free Survival. Female. Germany / epidemiology. Humans. Infant. Infant, Newborn. Italy / epidemiology. Male. Multivariate Analysis. Proportional Hazards Models. Risk Factors. Survival Rate. Treatment Outcome

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  • [ErratumIn] J Clin Oncol. 2006 Feb 1;24(4):724. Koscielniak, Eura [corrected to Koscielniak, Ewa]
  • (PMID = 16293873.001).
  • [ISSN] 0732-183X
  • [Journal-full-title] Journal of clinical oncology : official journal of the American Society of Clinical Oncology
  • [ISO-abbreviation] J. Clin. Oncol.
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article; Multicenter Study; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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66. Bisogno G, Sotti G, Nowicki Y, Ferrari A, Garaventa A, Zanetti I, Favre C, Schiavetti A, Tamaro P, Carli M: Soft tissue sarcoma as a second malignant neoplasm in the pediatric age group. Cancer; 2004 Apr 15;100(8):1758-65
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  • [Title] Soft tissue sarcoma as a second malignant neoplasm in the pediatric age group.
  • BACKGROUND: Survivors of childhood malignancies have an increased risk of developing second malignant neoplasms (SMN) due to their prior treatment and/or genetic susceptibility.
  • A small proportion of SMNs are soft tissue sarcomas (STS), whose prognosis is generally thought to be poor, though publications on such patients' treatment and outcome is limited.
  • The primary tumor was STS in five patients; Hodgkin disease in five patients; leukemia in four patients; retinoblastoma, neuroblastoma, and Wilms tumor in two patients each; and other tumor types in five patients.
  • SMNs occurred after a median of 8 years (range, 1.9-15.0 years) and included rhabdomyosarcoma (RMS) in 4 patients, malignant peripheral nerve sheath tumor in 4 patients, extraosseous Ewing family tumor (EFT) in 4 patients, leiomyosarcoma in 3 patients, fibrosarcoma in 2 patients, synovial sarcoma in 2 patients, and other tumor types in 6 patients.
  • Treatment generally was administered according to the guidelines for primary STS.
  • RESULTS: Seven non-RMS patients with STS underwent surgery alone, whereas 18 patients received chemotherapy and 8 patients received radiotherapy.
  • Fifteen patients were alive in complete remission of their SMN at the time of last follow-up.
  • Responses to chemotherapy and survival were satisfactory for patients with tumors such as RMS and EFT.
  • Complete tumor resection was correlated with a favorable prognosis in patients with other types of STS and in patients with postirradiation sarcoma.
  • Two patients developed a third malignancy.
  • CONCLUSIONS: Although prior treatment may hinder the management of these patients, pediatric STS second malignancies can be cured using the same strategies used for de novo pediatric sarcomas.
  • Long-term follow-up is mandatory given the risks of further malignancies and more severe, treatment-related side effects.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Neoplasms, Second Primary. Sarcoma / surgery. Soft Tissue Neoplasms / surgery
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Combined Modality Therapy. Female. Follow-Up Studies. Humans. Male. Prognosis. Survival Analysis. Treatment Outcome

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  • [Copyright] Copyright 2004 American Cancer Society.
  • (PMID = 15073867.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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67. Sierszeń W, Stankiewicz C: [Malignant schwannoma of infraorbital nerve]. Otolaryngol Pol; 2003;57(4):573-6
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  • [Title] [Malignant schwannoma of infraorbital nerve].
  • Schwannoma of the head and neck may develop from many nerves, i.e. cranial or spinal.
  • The authors described malignant schwannoma of infraorbital nerve.
  • Patient was treated by combined method: in first step chemotherapy, in second surgical excision with 2 year disease-free survival.
  • [MeSH-major] Neurilemmoma / pathology. Oculomotor Nerve / pathology. Peripheral Nervous System Neoplasms / pathology

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  • (PMID = 14587398.001).
  • [ISSN] 0030-6657
  • [Journal-full-title] Otolaryngologia polska = The Polish otolaryngology
  • [ISO-abbreviation] Otolaryngol Pol
  • [Language] pol
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Poland
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68. Mrugala MM, Batchelor TT, Plotkin SR: Peripheral and cranial nerve sheath tumors. Curr Opin Neurol; 2005 Oct;18(5):604-10
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  • [Title] Peripheral and cranial nerve sheath tumors.
  • PURPOSE OF REVIEW: The intention of the authors is to provide the reader with an overview of the recent advances in the diagnosis and treatment of nerve sheath tumors.
  • Vestibular schwannomas, neurogenetic syndromes such as schwannomatosis and multiple isolated neurofibromas, and malignant peripheral nerve sheath tumors are covered in this review.
  • RECENT FINDINGS: Over the last year, literature focusing on different management strategies for patients with vestibular schwannomas dominated the field.
  • New insights into the biology of peripheral nerve tumor development and growth, including expression of vascular endothelial growth factor by vestibular schwannomas and the role of Notch signaling in malignant transformation of benign neurofibromas have been described.
  • Diagnostic criteria for schwannomatosis, a recently described condition, are being developed.
  • SUMMARY: Peripheral nerve tumors are classified according to the specific features of cellular differentiation.
  • The most common types include schwannoma and neurofibroma.
  • These tumors can occur sporadically or as manifestations of genetic syndromes such as neurofibromatosis types 1 and 2 or schwannomatosis.
  • The majority of peripheral nerve tumors are benign but malignant transformation does occur.
  • Metastatic tumors can also affect peripheral nerves.
  • Positron emission tomography is a useful technique in the presurgical differentiation between benign and malignant peripheral nerve sheath tumors.
  • Treatment is directed towards symptomatic control.
  • Surgery, radiation and, in rare instances, chemotherapy are the major treatment modalities employed.
  • [MeSH-major] Cranial Nerve Neoplasms. Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / therapy. Peripheral Nervous System Neoplasms

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  • (PMID = 16155448.001).
  • [ISSN] 1350-7540
  • [Journal-full-title] Current opinion in neurology
  • [ISO-abbreviation] Curr. Opin. Neurol.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] England
  • [Number-of-references] 33
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69. Garaventa A, Gambini C, Villavecchia G, Di Cataldo A, Bertolazzi L, Pizzitola MR, De Bernardi B, Haupt R: Second malignancies in children with neuroblastoma after combined treatment with 131I-metaiodobenzylguanidine. Cancer; 2003 Mar 1;97(5):1332-8
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  • [Title] Second malignancies in children with neuroblastoma after combined treatment with 131I-metaiodobenzylguanidine.
  • BACKGROUND: (131)I-metaiodobenzylguanidine ((131)I-MIBG) is selectively taken up by cells of neural crest origin, allowing targeted radiotherapy of tumors such as neuroblastoma (NB) and pheochromocytoma.
  • Radiotherapy may provide additional benefits in the treatment of NB, with moderate side effects such as hematologic and thyroid toxicity.
  • We describe our experience with second cancers occurring in children treated with (131)I-MIBG and chemotherapy.
  • METHODS: The clinical records of 119 consecutive NB cases treated with (131)I-MIBG at a single institution between 1984 and 2001 were reviewed for the occurrence of a second malignant neoplasm (SMN).
  • In particular, two cases of myeloid leukemia, one of angiomatous fibrous histiocytoma, one of malignant schwannoma, and one case of rhabdomyosarcoma were detected.
  • The schwannoma and the rhabdomyosarcoma developed within the residual neuroblastic mass after first-line therapy.
  • CONCLUSIONS: Should (131)I-MIBG treatment become more broadly employed in the therapeutic strategy for neuroblastoma, the risk of second cancer will have to be taken into consideration.
  • [MeSH-major] 3-Iodobenzylguanidine / therapeutic use. Neoplasms, Radiation-Induced. Neoplasms, Second Primary. Neuroblastoma / radiotherapy. Peripheral Nervous System Neoplasms / radiotherapy. Radiopharmaceuticals / therapeutic use
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Hypothyroidism / etiology. Infant. Male. Survival Rate. Treatment Outcome

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  • [Copyright] Copyright 2003 American Cancer Society.
  • (PMID = 12599242.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Radiopharmaceuticals; 35MRW7B4AD / 3-Iodobenzylguanidine
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70. Lambrou NC, Mirhashemi R, Wolfson A, Thesiger P, Penalver M: Malignant peripheral nerve sheath tumor of the vulva: a multimodal treatment approach. Gynecol Oncol; 2002 May;85(2):365-71
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  • [Title] Malignant peripheral nerve sheath tumor of the vulva: a multimodal treatment approach.
  • BACKGROUND: Malignant peripheral nerve sheath tumors (MPNSTs) are rare in the gynecological population and have a high risk for local and distant failures.
  • Multimodal management of a patient with MPNST of the vulva and review of the literature are outlined.
  • CASE: A 34-year-old woman presented with a complaint of a rapidly increasing pelvic mass, pain, and difficulty ambulating.
  • A disfiguring 20 x 20-cm vulvar mass was identified and a recurrent MPNST diagnosed.
  • Therapy included external-beam radiation, anterior pelvic exenteration with pelvic reconstruction, and adjuvant chemotherapy without complication.
  • CONCLUSION: It is recommended that for malignant peripheral nerve sheath tumors of the vulva, complete surgical resection be performed with adjuvant radiation and chemotherapy in selected cases.
  • [MeSH-major] Nerve Sheath Neoplasms / therapy. Vulvar Neoplasms / therapy
  • [MeSH-minor] Adult. Chemotherapy, Adjuvant. Female. Humans. Neurofibromatosis 1 / complications. Radiotherapy, Adjuvant

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  • [Copyright] (c) 2002 Elsevier Science (USA).
  • (PMID = 11972402.001).
  • [ISSN] 0090-8258
  • [Journal-full-title] Gynecologic oncology
  • [ISO-abbreviation] Gynecol. Oncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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71. Ilgner J, Rojas W, Biesterfeld S, Schürmann K, Zimny M, Westhofen M: [Low-grade malignant peripheral nerve sheath tumor of the neck soft tissues]. Laryngorhinootologie; 2001 Jan;80(1):39-42
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  • [Title] [Low-grade malignant peripheral nerve sheath tumor of the neck soft tissues].
  • [Transliterated title] Niedrig-maligner peripherer Nervenscheidentumor der Halsweichteile.
  • BACKGROUND: Malignant Peripheral Nerve Sheath Tumours (MPNST) either grow sporadically, after radiation or chemotherapy respectively.
  • Because of the multiform histologic picture they are often difficult to differentiate from other soft tissue tumours.
  • PATIENT: We present the case of a sporadic MPNST which developed from the vagus nerve of a 39-year-old patient following radiation of the neck 7 years before.
  • RESULTS AND CONCLUSIONS: Sporadic MPNST of the head and neck are comparatively rare.
  • With regard to the strong association with Neurofibromatosis I and the difficult differential diagnosis to other soft tissue tumours the emphasis should be put on excluding further manifestations of Neurofibromatosis I and of secondary tumours.
  • [MeSH-major] Cranial Nerve Neoplasms / diagnosis. Head and Neck Neoplasms / diagnosis. Neoplasms, Radiation-Induced / diagnosis. Nerve Sheath Neoplasms / diagnosis. Soft Tissue Neoplasms / diagnosis. Vagus Nerve Diseases / diagnosis
  • [MeSH-minor] Adult. Cell Transformation, Neoplastic / pathology. Diagnosis, Differential. Female. Humans. Neurofibromatosis 1 / diagnosis. Neurofibromatosis 1 / pathology. Neurofibromatosis 1 / surgery. Vagus Nerve / pathology

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  • (PMID = 11272246.001).
  • [ISSN] 0935-8943
  • [Journal-full-title] Laryngo- rhino- otologie
  • [ISO-abbreviation] Laryngorhinootologie
  • [Language] ger
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Germany
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72. Ferrari A, Bisogno G, Macaluso A, Casanova M, D'Angelo P, Pierani P, Zanetti I, Alaggio R, Cecchetto G, Carli M: Soft-tissue sarcomas in children and adolescents with neurofibromatosis type 1. Cancer; 2007 Apr 1;109(7):1406-12
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Soft-tissue sarcomas in children and adolescents with neurofibromatosis type 1.
  • BACKGROUND: Patients affected by neurofibromatosis type 1 (NF1) are at higher risk of developing soft-tissue sarcomas (STS) than the general population.
  • METHODS: The study included 37 patients with neurogenic sarcomas (36 malignant peripheral nerve sheath tumors [MPNST], 1 triton tumor) and 6 cases of rhabdomyosarcoma (RMS).
  • The prevalence of NF1 observed during the study period was 43% in the MPNST population and 1% in the RMS group.
  • RESULTS: Most patients with neurogenic sarcomas had large, invasive tumors.
  • Two of 16 patients with evaluable disease responded to chemotherapy.
  • All 6 RMS patients were </=3 years old and had embryonal subtype, 5 of 6 arising in the genitourinary tract or pelvis (paravesical); 4 were alive in first remission at the time of the analysis, 1 was alive in second remission after a local recurrence, and 1 died of disease.
  • CONCLUSIONS: The occurrence of STS in pediatric patients with NF1 syndrome in Italy is discussed, confirming that NF1 patients have a high risk of developing STS, and particularly MPNST, often with an aggressive clinical presentation and poor outcome.
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Infant. Male. Neoplasms, Second Primary / complications. Neoplasms, Second Primary / diagnosis. Neoplasms, Second Primary / therapy. Nerve Sheath Neoplasms / complications. Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / therapy. Neurofibrosarcoma / complications. Neurofibrosarcoma / diagnosis. Neurofibrosarcoma / therapy. Peripheral Nervous System Neoplasms / complications. Peripheral Nervous System Neoplasms / diagnosis. Peripheral Nervous System Neoplasms / therapy. Prognosis. Prospective Studies. Rhabdomyosarcoma / complications. Rhabdomyosarcoma / diagnosis. Rhabdomyosarcoma / therapy. Risk Factors. Survival Rate


73. Pirayesh A, Chee Y, Helliwell TR, Hershman MJ, Leinster SJ, Fordham MV, Poston GJ: The management of retroperitoneal soft tissue sarcoma: a single institution experience with a review of the literature. Eur J Surg Oncol; 2001 Aug;27(5):491-7
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  • [Title] The management of retroperitoneal soft tissue sarcoma: a single institution experience with a review of the literature.
  • AIM: Ten percent of soft tissue sarcomas (STS) arise in the retroperitoneal tissues.
  • The prognosis for patients with retroperitoneal sarcoma is poor with a 5-year survival rate between 12% and 70%.
  • Stage at presentation, high histological grade, unresectable primary tumour and incomplete resection are associated with a less favourable outcome.
  • Patient, tumour and treatment variables were analysed including use of adjuvant therapy and survival status.
  • Thirteen patients presented with tumours larger than 10 cm.
  • The tumours were seven liposarcomas, six leiomyosarcomas, three malignant fibrous histiocytomas, two rhabdomyosarcomas, two malignant schwannomas and two undifferentiated sarcomas.
  • Six primary tumours were completely excised, five patients received radiotherapy and five received chemotherapy.
  • Five patients received radiotherapy and five received chemotherapy.
  • The median survival for patients with primary tumours was 36 months, and 5-year survival was 44%.
  • Adjuvant therapy was not associated with higher survival rates.
  • Adjuvant radiotherapy and chemotherapy do not appear to be any proven benefit and the single most important prognostic factor is aggressive successful en bloc resection of the primary tumour.
  • [MeSH-major] Retroperitoneal Neoplasms / surgery. Soft Tissue Neoplasms / surgery
  • [MeSH-minor] Adult. Aged. Aged, 80 and over. Female. Follow-Up Studies. Histiocytoma, Benign Fibrous / surgery. Humans. Male. Middle Aged. Neurilemmoma / surgery. Prognosis. Retrospective Studies. Sarcoma / surgery. Survival Analysis. Tomography, X-Ray Computed. Treatment Outcome

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  • [Copyright] Copyright Harcourt Publishers Limited.
  • (PMID = 11504522.001).
  • [ISSN] 0748-7983
  • [Journal-full-title] European journal of surgical oncology : the journal of the European Society of Surgical Oncology and the British Association of Surgical Oncology
  • [ISO-abbreviation] Eur J Surg Oncol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] England
  • [Number-of-references] 53
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74. Mosca F, Stracqualursi A, Lipari G, Latteri F, Palazzo F, Russo G: [Malignant schwannoma of the small intestine: a report of 2 cases]. G Chir; 2000 Apr;21(4):149-55
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  • [Title] [Malignant schwannoma of the small intestine: a report of 2 cases].
  • [Transliterated title] Lo schwannoma maligno dell'intestino tenue: descrizione di due casi.
  • The authors report two cases of small bowel malignant schwannoma.
  • Histologic differentiation from other stromal tumors may require electron microscopy, although the preparation of immunohistochemical reactions now allows to identify forms which were previously diagnosed in different terms to be precisely classified.
  • Diagnosis is often late and no preoperative test enables a correct clinical diagnosis to be made.
  • The primary treatment is surgical and the resection is the only real therapy.
  • Radiotherapy and chemotherapy are ineffective.
  • [MeSH-major] Ileal Neoplasms / diagnosis. Ileocecal Valve. Jejunal Neoplasms / diagnosis. Neurilemmoma / diagnosis

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  • (PMID = 10812769.001).
  • [ISSN] 0391-9005
  • [Journal-full-title] Il Giornale di chirurgia
  • [ISO-abbreviation] G Chir
  • [Language] ita
  • [Publication-type] Case Reports; English Abstract; Journal Article; Review
  • [Publication-country] ITALY
  • [Number-of-references] 19
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75. Sar C, Eralp L: Metastatic spinal neurofibrosarcoma. Arch Orthop Trauma Surg; 2002 Mar;122(2):106-8
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  • [Title] Metastatic spinal neurofibrosarcoma.
  • Neurofibrosarcomas are rare tumors usually arising in somatic soft tissues or peripheral nerves.
  • Four cases of metastatic neurofibrosarcoma to the spine have been reported before.
  • A 30-year-old woman with neurofibromatosis and a history of previous neurofibrosarcoma resection presented with back pain.
  • After the completion of adjuvant chemotherapy, a solitary pulmonary nodule was detected.
  • Though local control can be achieved in more than 80% of the patients with neurofibrosarcoma by wide surgical resection followed by adjuvant chemo- and radiotherapy, most patients die of systemic metastasis.
  • The current patient survived 50 months after the initial resection of a forearm neurofibrosarcoma.
  • Prolonged survival with the help of chemo- and radiotherapy justifies our aggressive surgical strategy for the treatment of spinal metastasis in order to achieve neurologic cure and spinal stability.
  • [MeSH-major] Neurofibrosarcoma / secondary. Spinal Neoplasms / secondary. Spinal Neoplasms / therapy
  • [MeSH-minor] Adult. Biopsy, Needle. Bone Neoplasms / pathology. Bone Neoplasms / therapy. Chemotherapy, Adjuvant. Combined Modality Therapy. Female. Follow-Up Studies. Forearm. Humans. Immunohistochemistry. Magnetic Resonance Imaging. Radiotherapy, Adjuvant. Spinal Fusion. Treatment Outcome

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  • (PMID = 11880913.001).
  • [ISSN] 0936-8051
  • [Journal-full-title] Archives of orthopaedic and trauma surgery
  • [ISO-abbreviation] Arch Orthop Trauma Surg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
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76. Ishimaru A, Hasegawa J, Shinbo K, Arahara T, Kinoshita Y, Shimoyama E, Korehisa M, Oonuki M, Miyazawa T, Itabashi M: [A case of retroperitoneal tumor successfully resected thanks to effective chemotherapy]. Gan To Kagaku Ryoho; 2009 Jun;36(6):1007-11
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  • [Title] [A case of retroperitoneal tumor successfully resected thanks to effective chemotherapy].
  • First, pathological diagnosis of sarcoma was made by fine needle aspiration, then secondary pathological diagnosis of suspected malignant schwannoma was made by a partial resection.
  • The final pathological diagnosis by operation was immature teratoma with embryonal carcinoma.
  • The preoperative diagnosis was difficult in this case, and the tumor had grown too large after partial resection to surgically resect.
  • The effective chemotherapy reduced this tumor enough to allow successful resection.
  • [MeSH-minor] Carcinoma, Embryonal / drug therapy. Carcinoma, Embryonal / pathology. Carcinoma, Embryonal / surgery. Humans. Male. Young Adult

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  • (PMID = 19542726.001).
  • [ISSN] 0385-0684
  • [Journal-full-title] Gan to kagaku ryoho. Cancer & chemotherapy
  • [ISO-abbreviation] Gan To Kagaku Ryoho
  • [Language] jpn
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Japan
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77. Komdeur R, Plaat BE, van der Graaf WT, Hoekstra HJ, Hollema H, van den Berg E, Zwart N, Scheper RJ, Molenaar WM: Expression of multidrug resistance proteins, P-gp, MRP1 and LRP, in soft tissue sarcomas analysed according to their histological type and grade. Eur J Cancer; 2003 May;39(7):909-16
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Expression of multidrug resistance proteins, P-gp, MRP1 and LRP, in soft tissue sarcomas analysed according to their histological type and grade.
  • The biological behaviour of different histological types and grades of soft tissue sarcomas (STS) varies.
  • This might result in a differing sensitivity to cytotoxic drugs.
  • Cross-resistance to functionally and structurally distinct natural-product drugs, known as multidrug resistance (MDR), is associated with the overexpression of P-glycoprotein (P-gp), multidrug resistance-associated protein 1 (MRP1) and lung resistance-related protein (LRP).
  • The purpose of this study was to evaluate the expression of P-gp, MRP1 and LRP in STS according to their histological type and grade.
  • In 141 chemotherapy-naive STS patients, the expression of the three MDR proteins was detected by immunohistochemistry.
  • Nine histological types were documented.
  • These were 19% grade 1, 34% grade 2 and 47% grade 3 tumours.
  • Expression of P-gp and LRP was observed more frequently than the expression of MRP1 (P<0.0001).
  • P-gp expression was most pronounced in malignant fibrous histiocytoma (MFH), but was low in leiomyosarcomas.
  • MRP1 was expressed in most malignant peripheral nerve sheath tumours (MPNST).
  • MRP1 and LRP expression was significantly more common in grades 2 and 3 compared with grade 1 tumours.
  • In conclusion, P-gp, MRP1 and LRP are expressed in the majority of STS, but this expression varies according to the histological type.
  • MRP1 and LRP, but not P-gp expression, were found to be correlated to tumour grade.
  • [MeSH-major] Multidrug Resistance-Associated Proteins / metabolism. Neoplasm Proteins / metabolism. P-Glycoproteins / metabolism. Sarcoma / drug therapy. Soft Tissue Neoplasms / drug therapy. Vault Ribonucleoprotein Particles / metabolism
  • [MeSH-minor] Adolescent. Adult. Aged. Aged, 80 and over. Child. Child, Preschool. Drug Resistance, Multiple. Drug Resistance, Neoplasm. Female. Humans. Immunohistochemistry / methods. Infant. Male. Middle Aged

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  • (PMID = 12706359.001).
  • [ISSN] 0959-8049
  • [Journal-full-title] European journal of cancer (Oxford, England : 1990)
  • [ISO-abbreviation] Eur. J. Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] England
  • [Chemical-registry-number] 0 / ABCC4 protein, human; 0 / Multidrug Resistance-Associated Proteins; 0 / Neoplasm Proteins; 0 / P-Glycoproteins; 0 / Vault Ribonucleoprotein Particles; 0 / major vault protein
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78. Moharir M, London K, Howman-Giles R, North K: Utility of positron emission tomography for tumour surveillance in children with neurofibromatosis type 1. Eur J Nucl Med Mol Imaging; 2010 Jul;37(7):1309-17
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  • [Title] Utility of positron emission tomography for tumour surveillance in children with neurofibromatosis type 1.
  • PURPOSE: There is little consensus regarding optimal surveillance of optic pathway glioma (OPG) and plexiform neurofibroma (PNF) in childhood neurofibromatosis type 1 (NF1). (18)F-2-Fluoro-2-deoxy-D: -glucose (FDG) positron emission tomography and computed tomography (PET/CT) is employed in the surveillance of adult PNFs; but its utility has neither been specifically studied in children with PNFs nor in children with OPG.
  • FDG-avidity of tumours was semi-quantitatively analysed and graded by calculating the maximum standardised uptake value (SUV(max)) [grade 1: <3 (low), grade 2: >3-<4 (intermediate), grade 3: >4 (intense)].
  • FDG-avidity reduced from grade 3 to grade 1 in two symptomatic OPGs following chemotherapy and this was associated with clinical improvement.
  • PET/CT diagnosed symptomatic OPGs with a sensitivity of 0.625 [95% confidence interval (CI): 0.259-0.897] and specificity of 0.875 (95% CI: 0.466-0.993).
  • The two grade 3 PNFs were confirmed malignant peripheral nerve sheath tumours.
  • PET/CT diagnosed malignant transformation with a sensitivity of 1.0 (95% CI: 0.197-1.0) and specificity of 0.857 (95% CI: 0.561-0.974).
  • CONCLUSION: PET/CT may contribute useful information to the surveillance of OPG in childhood NF1-particularly to identify progressive, symptomatic tumours.
  • As in adults, PET/CT is useful for the detection of malignant transformation in PNFs in children with NF1.
  • [MeSH-major] Neurofibromatosis 1 / diagnostic imaging. Positron-Emission Tomography
  • [MeSH-minor] Adult. Child. Child, Preschool. Female. Fluorodeoxyglucose F18. Humans. Male. Neurofibroma, Plexiform / diagnostic imaging. Optic Nerve Glioma / diagnostic imaging. Retrospective Studies. Tomography, X-Ray Computed

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  • (PMID = 20179923.001).
  • [ISSN] 1619-7089
  • [Journal-full-title] European journal of nuclear medicine and molecular imaging
  • [ISO-abbreviation] Eur. J. Nucl. Med. Mol. Imaging
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0Z5B2CJX4D / Fluorodeoxyglucose F18
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79. Qi X, Chu Z, Mahller YY, Stringer KF, Witte DP, Cripe TP: Cancer-selective targeting and cytotoxicity by liposomal-coupled lysosomal saposin C protein. Clin Cancer Res; 2009 Sep 15;15(18):5840-51
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  • EXPERIMENTAL DESIGN: Neuroblastoma, malignant peripheral nerve sheath tumor and, breast cancer cells were treated with saposin C-dioleoylphosphatidylserine nanovesicles and assessed for cell viability, ceramide elevation, caspase activation, and apoptosis.
  • Fluorescently labeled saposin C-dioleoylphosphatidylserine was i.v. injected to determine in vivo tumor-targeting specificity.
  • RESULTS: Saposin C-dioleoylphosphatidylserine nanovesicles, with a mean diameter of approximately 190 nm, showed specific tumor-targeting activity shown through in vivo imaging.
  • Following i.v. administration, saposin C-dioleoylphosphatidylserine nanovesicles preferentially accumulated in tumor vessels and cells in tumor-bearing mice.
  • Saposin C-dioleoylphosphatidylserine induced apoptosis in multiple cancer cell types while sparing normal cells and tissues.
  • In in vivo models, saposin C-dioleoylphosphatidylserine nanovesicles significantly inhibited growth of preclinical xenografts of neuroblastoma and malignant peripheral nerve sheath tumor. I.v. dosing of saposin C-dioleoylphosphatidylserine showed no toxic effects in nontumor tissues.
  • [MeSH-major] Antineoplastic Agents / pharmacology. Apoptosis / drug effects. Lysosomes / chemistry. Neoplasms / drug therapy. Phosphatidylserines / chemistry. Saposins / pharmacology. Saposins / therapeutic use
  • [MeSH-minor] Animals. Breast Neoplasms / drug therapy. Breast Neoplasms / pathology. Cell Proliferation / drug effects. Disease Models, Animal. Drug Screening Assays, Antitumor. Drug-Related Side Effects and Adverse Reactions. Female. Humans. Liposomes. Mice. Nerve Sheath Neoplasms / drug therapy. Nerve Sheath Neoplasms / pathology. Neuroblastoma / drug therapy. Neuroblastoma / pathology. Substrate Specificity. Tumor Cells, Cultured

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  • (PMID = 19737950.001).
  • [ISSN] 1078-0432
  • [Journal-full-title] Clinical cancer research : an official journal of the American Association for Cancer Research
  • [ISO-abbreviation] Clin. Cancer Res.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Liposomes; 0 / Phosphatidylserines; 0 / Saposins; 70614-14-1 / 1,2-dioleoylphosphatidylserine
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80. Minovi A, Basten O, Hunter B, Draf W, Bockmühl U: Malignant peripheral nerve sheath tumors of the head and neck: management of 10 cases and literature review. Head Neck; 2007 May;29(5):439-45
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  • [Title] Malignant peripheral nerve sheath tumors of the head and neck: management of 10 cases and literature review.
  • BACKGROUND: This study analyzes the management and outcomes of a series of 10 malignant peripheral nerve sheath tumors (MPNST) of the head and neck.
  • METHODS: From 1984 to 2004, 10 patients underwent surgical treatment of a MPNST.
  • RESULTS: Eight tumors were located at the lateral skull base; 2 involved the vagus nerve in isolation.
  • Seventy percent of the tumors could be resected completely.
  • Negative prognostic indicators were advanced tumor stage, early recurrence, and presumably also the presence of von Recklinghausen's disease.
  • CONCLUSIONS: Although rare, MPNST is one of the most aggressive tumors in the head and neck area.
  • Complete tumor removal is the mainstay of treatment and most important prognostic factor of MPNST.
  • The role of adjuvant chemotherapy remains controversial.
  • [MeSH-major] Head and Neck Neoplasms / mortality. Head and Neck Neoplasms / therapy. Neoplasm Recurrence, Local / mortality. Nerve Sheath Neoplasms / mortality. Nerve Sheath Neoplasms / therapy
  • [MeSH-minor] Adolescent. Adult. Aged. Chemotherapy, Adjuvant. Female. Humans. Male. Middle Aged. Prognosis. Radiotherapy, Adjuvant. Retrospective Studies. Survival Rate

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  • [Copyright] (c) 2006 Wiley Periodicals, Inc.
  • (PMID = 17163467.001).
  • [ISSN] 1043-3074
  • [Journal-full-title] Head & neck
  • [ISO-abbreviation] Head Neck
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 34
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81. Dobashi Y, Suzuki S, Sato E, Hamada Y, Yanagawa T, Ooi A: EGFR-dependent and independent activation of Akt/mTOR cascade in bone and soft tissue tumors. Mod Pathol; 2009 Oct;22(10):1328-40
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  • [Title] EGFR-dependent and independent activation of Akt/mTOR cascade in bone and soft tissue tumors.
  • To gain the insight into the involvement of signaling mediated by the mammalian target of rapamycin (mTOR) in the phenotype and biological profiles of tumors and tumor-like lesions of the bone and soft tissue, we analyzed the expression and phosphorylation (activation) of mTOR and its correlation with the status of upstream and downstream modulator proteins Akt, p70S6-kinase (S6K), and eukaryotic initiation factor 4E-binding protein 1 (4E-BP1), which we refer to collectively as mTOR cassette proteins.
  • Immunohistochemical analysis of 140 cases showed activation of Akt in 55% (61% in malignant and 27% in benign), and mTOR expression in 61% (66% in malignant and 39% in benign).
  • The preponderance of mTOR activation was found in tumors of peripheral nerve sheath (malignant peripheral nerve sheath tumor and schwannoma), skeletal muscle origin (rhabdomyosarcoma), and in those exhibiting epithelial nature (chordoma and synovial sarcoma).
  • Together with the result of immunoblotting analysis, it was shown that many of those particular tumors with mTOR activation exhibited activation of Akt, S6K, and 4E-BP1, suggesting the constitutive activation of the Akt/mTOR pathway.
  • We conclude that mTOR-mediated signaling proteins function not only in the proliferation of the tumor cells, but also in the differentiation and/or maintenance of morphological phenotypes in tumors of rhabdomyoblastic and nerve sheath cell origin.
  • Furthermore, mTOR signaling may also modulate morphogenesis of tumors exhibiting epithelial nature.
  • Overall, these results suggest that inhibitors of mTOR cassette may be useful as novel components of combined chemotherapy for a defined subset of bone and soft tissue sarcomas.
  • [MeSH-major] Adaptor Proteins, Signal Transducing / analysis. Bone Neoplasms / enzymology. Phosphoproteins / analysis. Protein Kinases / analysis. Proto-Oncogene Proteins c-akt / analysis. Receptor, Epidermal Growth Factor / analysis. Ribosomal Protein S6 Kinases, 70-kDa / analysis. Signal Transduction. Soft Tissue Neoplasms / enzymology
  • [MeSH-minor] Cell Proliferation. Enzyme Activation. Humans. Immunoblotting. Immunohistochemistry. Mutation. Neoplasm Staging. Phosphorylation. Prognosis. TOR Serine-Threonine Kinases

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  • (PMID = 19648884.001).
  • [ISSN] 1530-0285
  • [Journal-full-title] Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc
  • [ISO-abbreviation] Mod. Pathol.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Adaptor Proteins, Signal Transducing; 0 / EIF4EBP1 protein, human; 0 / Phosphoproteins; EC 2.7.- / Protein Kinases; EC 2.7.1.1 / MTOR protein, human; EC 2.7.1.1 / TOR Serine-Threonine Kinases; EC 2.7.10.1 / EGFR protein, human; EC 2.7.10.1 / Receptor, Epidermal Growth Factor; EC 2.7.11.1 / Proto-Oncogene Proteins c-akt; EC 2.7.11.1 / Ribosomal Protein S6 Kinases, 70-kDa
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82. Rawal A, Yin Q, Roebuck M, Sinopidis C, Kalogrianitis S, Helliwell TR, Frostick S: Atypical and malignant peripheral nerve-sheath tumors of the brachial plexus: report of three cases and review of the literature. Microsurgery; 2006;26(2):80-6
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  • [Title] Atypical and malignant peripheral nerve-sheath tumors of the brachial plexus: report of three cases and review of the literature.
  • Tumor involvement of the brachial plexus is uncommon.
  • The most common intrinsic neoplasms involving the brachial plexus are benign neurilemmomas and neurofibromas that are usually associated with neurofibromatosis-1 (NF-1).
  • Malignant peripheral nerve-sheath tumors (MPNST) are rare at this site, arising spontaneously or in the context of NF-1.
  • This presentation discusses the clinical presentation, pathology, and management of these tumors, which usually occur in young adults.
  • MPNST are intermediate or high-grade sarcomas with a high risk of local and distant spread.
  • Approximately 50% of MPNST arise in patients with NF-1, and therefore these patients should be thoroughly investigated for any new symptoms or masses.
  • MPNST of the brachial plexus should be treated with an adequate wide local excision, with adjuvant high-dose radiotherapy pre- or postoperatively.
  • The role of chemotherapy in the treatment of MPNST is not clearly defined, but it may have some benefit in salvaging treatment failures.
  • [MeSH-major] Brachial Plexus. Nerve Sheath Neoplasms / pathology. Nerve Sheath Neoplasms / surgery

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  • (PMID = 16538633.001).
  • [ISSN] 0738-1085
  • [Journal-full-title] Microsurgery
  • [ISO-abbreviation] Microsurgery
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 24
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83. Ishihara S, Honda Y, Asato T, Nonaka M, Nakagawa S, Hirashima K, Hayashi N, Baba H, Iyama K: Interdigitating dendritic cell sarcoma of the ileum recurred in multiple lymph nodes and duodenum three years after operation without chemotherapy. Pathol Res Pract; 2010 Jul 15;206(7):514-8
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  • [Title] Interdigitating dendritic cell sarcoma of the ileum recurred in multiple lymph nodes and duodenum three years after operation without chemotherapy.
  • Here we describe a case of a 47-year-old man with interdigitating dendritic cell sarcoma (IDCS) in the ileum.
  • The ileal tumor, measuring 2cm, was detected and resected with regional lymphadenectomy.
  • At that time, a pathologic diagnosis of malignant peripheral nerve sheath tumor was made.
  • The patient, who was not treated with chemotherapy, showed no signs of recurrence.
  • Oval to spindle-shaped atypical cells, which resembled ileal tumor cells, infiltrated into the lymph node and duodenum.
  • Based on the histologic and immunohistochemical analysis, the histopathologic diagnosis of IDCS was confirmed.
  • To our knowledge, five cases of IDCS arising in the intestinal tract have been reported to date, and only one case, treated with both surgery and chemotherapy, led to remission.
  • This is the first case that has a comparatively favorable prognosis without chemotherapy after surgery.
  • [MeSH-major] Dendritic Cell Sarcoma, Interdigitating / pathology. Duodenal Neoplasms / pathology. Ileal Neoplasms / pathology. Lymph Nodes / pathology. Neoplasm Recurrence, Local / pathology
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Biomarkers, Tumor / analysis. Diagnostic Errors. Digestive System Surgical Procedures. Humans. Immunohistochemistry. Male. Middle Aged. Nerve Sheath Neoplasms / pathology

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  • [Copyright] Copyright 2010 Elsevier GmbH. All rights reserved.
  • (PMID = 20399026.001).
  • [ISSN] 1618-0631
  • [Journal-full-title] Pathology, research and practice
  • [ISO-abbreviation] Pathol. Res. Pract.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Biomarkers, Tumor
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84. Holtkamp N, Malzer E, Zietsch J, Okuducu AF, Mucha J, Mawrin C, Mautner VF, Schildhaus HU, von Deimling A: EGFR and erbB2 in malignant peripheral nerve sheath tumors and implications for targeted therapy. Neuro Oncol; 2008 Dec;10(6):946-57
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  • [Title] EGFR and erbB2 in malignant peripheral nerve sheath tumors and implications for targeted therapy.
  • Malignant peripheral nerve sheath tumors (MPNSTs) are sarcomas with poor prognosis and limited treatment options.
  • Evidence for a role of epidermal growth factor receptor (EGFR) and receptor tyrosine kinase erbB2 in MPNSTs led us to systematically study these potential therapeutic targets in a larger tumor panel (n = 37).
  • Multiplex ligation-dependent probe amplification and fluorescence in situ hybridization analysis revealed increased EGFR dosage in 28% of MPNSTs.
  • ERBB2 and three tumor suppressor genes (PTEN [phosphatase and tensin homolog deleted on chromosome 10], CDKN2A [cyclin-dependent kinase inhibitor 2A], and TP53 [tumor protein p53]) were frequently lost or reduced.
  • Comparison of corresponding neurofibromas and MPNSTs revealed an increase in genetic lesions in MPNSTs.
  • However, at the protein level, expression of EGFR and erbB2 was frequently detected in MPNSTs.
  • The EGFR ligands transforming growth factor alpha and EGF were more strongly expressed in MPNSTs than in neurofibromas.
  • The effects of the drugs erlotinib and trastuzumab, which target EGFR and erbB2, were determined on MPNST cell lines.
  • Summarized, our data indicate that EGFR and erbB2 are potential targets in treatment of MPNST patients.
  • [MeSH-major] Antineoplastic Agents / administration & dosage. Drug Delivery Systems. Nerve Sheath Neoplasms / genetics. Receptor, Epidermal Growth Factor / genetics. Receptor, ErbB-2 / genetics
  • [MeSH-minor] Antibodies, Monoclonal / pharmacology. Antibodies, Monoclonal, Humanized. Blotting, Western. Cell Line, Tumor. Cell Proliferation / drug effects. Drug Synergism. Epidermal Growth Factor / genetics. Epidermal Growth Factor / metabolism. Erlotinib Hydrochloride. Gene Dosage. Genes, p16. Genes, p53. Humans. Immunohistochemistry. PTEN Phosphohydrolase / genetics. Polymorphism, Single-Stranded Conformational. Quinazolines / pharmacology. Reverse Transcriptase Polymerase Chain Reaction. Transforming Growth Factor alpha / genetics. Transforming Growth Factor alpha / metabolism. Trastuzumab


85. Ploegmakers MJ, Pruszczynski M, De Rooy J, Kusters B, Veth RP: Angiosarcoma with malignant peripheral nerve sheath tumour developing in a patient with klippel-trénaunay-weber syndrome. Sarcoma; 2005;9(3-4):137-40
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  • [Title] Angiosarcoma with malignant peripheral nerve sheath tumour developing in a patient with klippel-trénaunay-weber syndrome.
  • PURPOSE: We discuss the coexistence of Klippel-Trénaunay-Weber syndrome with various malignancies, the possible histogenetic pathways and therapeutic implications.
  • METHODS: Our patient underwent an above-knee amputation for biopsy-proven malignant vascular tumour, first thought to be a composite hemangio-endothelioma and/or angiosarcoma with lung metastases.
  • RESULTS: In the amputated extremity, a vascular malformation was found with tumour showing various components with foci of angiosarcoma adjacent to diffuse neurofibroma and areas with high-grade malignant peripheral nerve sheath tumour.
  • Amputation and palliative chemotherapy were indicated, but he died of pulmonary and cerebral metastases 2 months postoperatively.
  • DISCUSSION: This case describes an angiosarcoma with malignant peripheral nerve sheath tumour developing in a patient with Klippel-Trénaunay-Weber syndrome.

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  • (PMID = 18521421.001).
  • [ISSN] 1357-714X
  • [Journal-full-title] Sarcoma
  • [ISO-abbreviation] Sarcoma
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Egypt
  • [Other-IDs] NLM/ PMC2395629
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86. Jankulovski N, Stankov O, Banev S, Petrovski D, Mickovski A, Mitevski A, Filipovski V, Popov Z: Isolated malignant peripheral nerve sheath tumor of kidney capsule. Prilozi; 2008 Dec;29(2):361-69
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  • [Title] Isolated malignant peripheral nerve sheath tumor of kidney capsule.
  • The occurrence of an isolated malignant peripheral nerve sheath tumor (MPNST) of the kidney capsule is extremely rare and its presence may only be expressed by an insidious onset of non-specific and misleading symptoms with a predominance of lower back pain.
  • A computer tomography (CT) scan (as the imaging procedure of choice) will demonstrate the tumor location and its relation to the surrounding structures.
  • Tumor excision in toto is considered the treatment of choice, but it can be hazardous, especially if the tumor is adhering to the surrounding structures.
  • If malignancy can safely be excluded, a laparoscopic excision should be considered as an alternative treatment as recurrence is unlikely.
  • Definition of the originating nerve might not always be possible, and a minor degree of neurological impairment has therefore to be anticipated.
  • A case of an isolated MPNST of the kidney capsule without neurofibromatosis is presented.
  • The tumor was located in the fatty and fibrous capsule.
  • The patient was further managed with radiotherapy and chemotherapy.
  • An MPNST in such a location is very unusual.


87. Kawai A, Kondo T, Suehara Y, Kikuta K, Hirohashi S: Global protein-expression analysis of bone and soft tissue sarcomas. Clin Orthop Relat Res; 2008 Sep;466(9):2099-106
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  • [Title] Global protein-expression analysis of bone and soft tissue sarcomas.
  • Analysis of global protein expression, an approach known as expression proteomics, can offer important clues for understanding tumor biology that cannot be obtained by other approaches (e.g., genome or transcriptome analysis).
  • Using two-dimensional difference gel electrophoresis (2D-DIGE) and mass spectrometry, we performed global protein expression studies of bone and soft tissue sarcomas to develop novel diagnostic and therapeutic biomarkers and allow molecular classification of the tumors.
  • Among 1500 protein variants identified in the two-dimensional gel, 67 proteins correctly distinguished the eight subtypes of 99 histologically classified soft tissue sarcomas.
  • Hierarchical clustering demonstrated leiomyosarcoma and MFH shared a similar protein expression profile, and clear cell sarcoma, synovial sarcoma, and MPNST could be grouped according to their protein expression patterns.
  • Patients with gastrointestinal stromal tumors expressing pfetin protein had better survival than those whose tumors lacked it.
  • We identified 10 protein spots associated with the chemosensitivity of osteosarcoma to preoperative chemotherapy.
  • These 10 spots could be new diagnostic and prognostic markers for osteosarcoma and new therapeutic targets for the disease.
  • Proteomic analysis using 2D-DIGE provides novel information on the biology of bone and soft tissue sarcomas that could be used to diagnosis and treat these tumors.
  • [MeSH-minor] Electrophoresis, Gel, Two-Dimensional. Gastrointestinal Stromal Tumors / metabolism. Gastrointestinal Stromal Tumors / mortality. Humans. Immunohistochemistry. Proportional Hazards Models. Proteins / metabolism. Proteomics

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  • (PMID = 18535868.001).
  • [ISSN] 1528-1132
  • [Journal-full-title] Clinical orthopaedics and related research
  • [ISO-abbreviation] Clin. Orthop. Relat. Res.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / KCTD12 protein, human; 0 / Proteins
  • [Other-IDs] NLM/ PMC2493021
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88. Hazarika P: Solitary malignant schwanoma of para pharyngeal space-a case report and review of literature. Indian J Otolaryngol Head Neck Surg; 2003 Oct;55(4):277-80
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  • [Title] Solitary malignant schwanoma of para pharyngeal space-a case report and review of literature.
  • Malignnant Schwannom is an aggressive tissue tumor that is more cnmitutnh found m assoiiatiun with Ion Recklinghausens disease.
  • The solitary tumors although rarer have a better prognosis when compared to those associated witk tan Recklinghausens disease.
  • Parupharyngeui tumors are rare and a majority af these are benign salivary or neurogenie tumors, A malignant Schwannoma at this site is very infrequent with only four earn reported so far.
  • The tase of a 16 year old girl with a parapharynccal malignant Schwnnaomu is presented Jor its rarity and our experience in dealing with it. .A combined modallty oftretument with surttery, radiotherapy and chemotherapy Was used.

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  • (PMID = 23120001.001).
  • [ISSN] 2231-3796
  • [Journal-full-title] Indian journal of otolaryngology and head and neck surgery : official publication of the Association of Otolaryngologists of India
  • [ISO-abbreviation] Indian J Otolaryngol Head Neck Surg
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] India
  • [Other-IDs] NLM/ PMC3451183
  • [Keywords] NOTNLM ; Malignant Schwannoma / Parapharyngeal Space Tumors
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89. Banerjee S, Byrd JN, Gianino SM, Harpstrite SE, Rodriguez FJ, Tuskan RG, Reilly KM, Piwnica-Worms DR, Gutmann DH: The neurofibromatosis type 1 tumor suppressor controls cell growth by regulating signal transducer and activator of transcription-3 activity in vitro and in vivo. Cancer Res; 2010 Feb 15;70(4):1356-66
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  • [Title] The neurofibromatosis type 1 tumor suppressor controls cell growth by regulating signal transducer and activator of transcription-3 activity in vitro and in vivo.
  • Neurofibromatosis type 1 (NF1) is a common cancer predisposition syndrome in which affected individuals develop benign and malignant nerve tumors.
  • The NF1 gene product neurofibromin negatively regulates Ras and mammalian target of rapamycin (mTOR) signaling, prompting clinical trials to evaluate the ability of Ras and mTOR pathway inhibitors to arrest NF1-associated tumor growth.
  • To discover other downstream targets of neurofibromin, we performed an unbiased cell-based high-throughput chemical library screen using NF1-deficient malignant peripheral nerve sheath tumor (MPNST) cells.
  • We further showed that signal transducer and activator of transcription-3 (STAT3), the target of cucurbitacin-I inhibition, was hyperactivated in NF1-deficient primary astrocytes and neural stem cells, mouse glioma cells, and human MPNST cells through Ser(727) phosphorylation, leading to increased cyclin D1 expression.
  • Finally, cucurbitacin-I inhibited the growth of NF1-deficient MPNST cells in vivo.
  • In summary, we used a chemical genetics approach to reveal STAT3 as a novel neurofibromin/mTOR pathway signaling molecule, define its action and regulation, and establish STAT3 as a tractable target for future NF1-associated cancer therapy studies.
  • [MeSH-minor] Animals. Animals, Newborn. Cells, Cultured. Chromones / pharmacology. Enzyme Inhibitors / pharmacology. Gene Expression Regulation / drug effects. Genes, Tumor Suppressor / physiology. Humans. Intracellular Signaling Peptides and Proteins / metabolism. Male. Mice. Mice, Nude. Mice, Transgenic. Morpholines / pharmacology. Protein-Serine-Threonine Kinases / metabolism. Signal Transduction / drug effects. Signal Transduction / genetics. TOR Serine-Threonine Kinases. Triterpenes / pharmacology. Xenograft Model Antitumor Assays


90. Charfeddine I, Mnejja M, Hammami B, Hasnaoui M, Hadj KA, Frikha I, Makni S, Boudawara T, Chakroun A, Ghorbel A: [Neurofibromatosis type 1 revealed by malignant peripheral nerve sheath tumor]. Rev Laryngol Otol Rhinol (Bord); 2009;130(4-5):327-30
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  • [Title] [Neurofibromatosis type 1 revealed by malignant peripheral nerve sheath tumor].
  • [Transliterated title] Tumeur maligne des gaines nerveuses périphériques révélant une neurofibromatose type 1.
  • Neurofibromatosis type 1 or Von Recklinghausen's disease is an affection with variable clinical expression.
  • Malignant transformation is rare and dangerous.
  • OBJECTIVE: The aim of this work is to study diagnostic criterias as well as treatment of this malignant transformation.
  • The histological exam concluded to a low grade malignant peripheral nerve sheath tumour Unfortunately despite a larger reoperation including resection of the manubrium, the limit of this excision were not safe.
  • Early recurrence was observed, although treated by surgery, chemotherapy and radiotherapy, the patient died by mediastinal invasion.
  • [MeSH-major] Nerve Sheath Neoplasms / pathology. Neurofibromatosis 1 / diagnosis. Soft Tissue Neoplasms / pathology


91. Munteanu M, Pîrşcoveanu M, Gugilă I, Munteanu MC, Munteanu AC, Ionescu M: [Rare case of retroperitoneal malignant Schwannoma]. Chirurgia (Bucur); 2004 Sep-Oct;99(5):345-50
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  • [Title] [Rare case of retroperitoneal malignant Schwannoma].
  • [Transliterated title] Caz rar de Schwannom malign retroperitoneal.
  • Malignant Schwannoma, recently renamed malignant peripheral nerve sheat tumor retroperitoneally localized, represents 0.01 of retroperitoneal tumours.
  • A 41-year old woman, without pathological record--and without cutaneous neurofibromatosis--hospitalized for increased volume of the abdomen, without symptomatology, is diagnosed after the imaging and biological tests--without CT and RMN--with retroperitoneal tumour.
  • The unusual size of tumour--6000 gr.
  • Two years after the operation, during which the patient was under chemotherapy, on a routine control we found a relapse at a distance from the initial tumour (primitive tumour ?
  • For the time being, after almost five years from the first operation, there are no clinical, biological and imaging changes.
  • CONCLUSIONS: the retroperitoneal space is quite enough for the development of large tumour masses, without symptomatology.
  • The present case combines most characteristics of retroperitoneal neoplasms: large or very large size, quasi-absent symptomatology, difficulty in preoperative diagnosis, surgical tactics and techniques--quite often, the total extirpation of tumour mass led to the sacrifice of other organs within the limits of a justified risk--and unforeseeable evolution with relapses having the same characteristics.
  • [MeSH-major] Neoplasm Recurrence, Local / therapy. Neurilemmoma / therapy. Retroperitoneal Neoplasms / therapy
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Chemotherapy, Adjuvant. Female. Humans. Pancreatectomy. Radiotherapy, Adjuvant. Splenectomy. Treatment Outcome

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  • (PMID = 15675290.001).
  • [ISSN] 1221-9118
  • [Journal-full-title] Chirurgia (Bucharest, Romania : 1990)
  • [ISO-abbreviation] Chirurgia (Bucur)
  • [Language] hun
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Romania
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92. Kochbati L, Boussen H, Benna F, Belhaj Ali Z, Gammoudi A, Bouaouina N, Besbes M, Ghilen L, Rahal K, Maalej M: [Second malignancies following Hodgkin's disease treatment in Tunisia. Retrospective study of 26 cases observed at the institute Salah-Azaïz]. Cancer Radiother; 2003 Oct;7(5):302-7
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  • [Title] [Second malignancies following Hodgkin's disease treatment in Tunisia. Retrospective study of 26 cases observed at the institute Salah-Azaïz].
  • [Transliterated title] Tumeurs secondaires après traitement pour maladie de Hodgkin en Tunisie. Etude rétrospective à propos de 26 cas observés à l'institut Salah-Azaïz.
  • METHODS AND PATIENTS: We consider as second cancer all tumours other than HD observed in patients after treatment for HD.
  • RESULTS: Twenty-five patients among 614 treated for HD between 1975 and 1991 developed 26 secondary tumours (4.2%).
  • Mean age at the diagnosis of HD was 32.5 years (12-56).
  • The first treatment was combined chemotherapy and radiotherapy in 22 cases and only chemotherapy in three cases (stage IV).
  • Mean dose was 41.3 Gy (2 Gy/fraction in 21 and 3.3 in one).
  • Chemotherapy was MOPP (13), MOPP and vinblastine (four), MOPP-ABVD (five), ABVD (two) and vinblastine only in one.
  • Mean delay of second tumours was 114.5 months (40-276).
  • There was five acute myeloid leukaemia, two digestive non-Hodgkin lymphomas, five nodal high-grade lymphomas, three breast cancers (one in man associated with thyroid cancer), five lung cancers (three non-small cell and two of small cell type), two gastric tumours and one rectal cancer, one synovialosarcoma of the knee and one malignant Schwannoma of the neck.
  • CONCLUSION: Second cancer risk after treatment for HD is not low.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / adverse effects. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Hodgkin Disease / drug therapy. Hodgkin Disease / radiotherapy. Neoplasms, Second Primary / epidemiology. Neoplasms, Second Primary / etiology

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  • (PMID = 14522350.001).
  • [ISSN] 1278-3218
  • [Journal-full-title] Cancer radiothérapie : journal de la Société française de radiothérapie oncologique
  • [ISO-abbreviation] Cancer Radiother
  • [Language] fre
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] France
  • [Chemical-registry-number] 11056-06-7 / Bleomycin; 35S93Y190K / Procarbazine; 50D9XSG0VR / Mechlorethamine; 5J49Q6B70F / Vincristine; 5V9KLZ54CY / Vinblastine; 7GR28W0FJI / Dacarbazine; 80168379AG / Doxorubicin; VB0R961HZT / Prednisone; ABVD protocol; MOPP protocol
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93. Bauer JA, Frye G, Bahr A, Gieg J, Brofman P: Anti-tumor effects of nitrosylcobalamin against spontaneous tumors in dogs. Invest New Drugs; 2010 Oct;28(5):694-702
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  • [Title] Anti-tumor effects of nitrosylcobalamin against spontaneous tumors in dogs.
  • PURPOSE: Given the limited options available to treat canine cancers, the use of companion animals for evaluating new drugs may identify better therapies for veterinary and human oncology.
  • The anti-tumor effects of nitrosylcobalamin (NO-Cbl), an apoptosis-inducing, vitamin B12-based carrier of nitric oxide (NO), was evaluated in four dogs with spontaneous cancer.
  • (1) A 13 year-old female spayed Giant Schnauzer with inoperable thyroid carcinoma and hypercalcemia. (2) A 6 year-old male neutered Golden Retriever with a malignant peripheral nerve sheath tumor (MPNST). (3) A ten yr-old neutered male Bichon Frise with apocrine gland anal sac adenocarcinoma (AGACA). (4) A 7 year-old female spayed Labrador mix with spinal meningioma following partial surgical resection.
  • Tumor regression was measured by physical exam and verified using ultrasound (case 1) and MRI (case 2-4).
  • (1) The Giant Schnauzer demonstrated a 77% reduction in tumor volume after ten weeks of daily NO-Cbl treatment. (2) The Golden Retriever demonstrated a 53% reduction in tumor volume after 15 months of daily NO-Cbl therapy. (3) The Bichon Frise demonstrated a 43% regression of the primary tumor and a 90% regression of an iliac lymph node measured by MRI after 15 months of treatment.
  • After 61 months, the dog currently has stable disease, normal liver enzymes, CBC analysis, and no evidence of toxicity. (4) The Labrador demonstrated complete regression of the residual tumor after 6 months of treatment.
  • CONCLUSION: We have shown previously that NO-Cbl is endocytosed by malignant cells, resulting in intra-tumoral NO release.
  • The use of NO-Cbl capitalizes on the tumor-specific properties of the vitamin B12 receptor and represents a promising anti-cancer therapy.
  • [MeSH-major] Dog Diseases / drug therapy. Neoplasms / veterinary. Nitroso Compounds / therapeutic use. Vitamin B 12 / analogs & derivatives
  • [MeSH-minor] Animals. Antineoplastic Agents / metabolism. Antineoplastic Agents / pharmacokinetics. Antineoplastic Agents / therapeutic use. Dogs. Dose-Response Relationship, Drug. Female. Magnetic Resonance Imaging. Male. Tumor Burden. Ultrasonography

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  • [ErratumIn] Invest New Drugs. 2011 Oct;29(5):1122
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  • (PMID = 19557306.001).
  • [ISSN] 1573-0646
  • [Journal-full-title] Investigational new drugs
  • [ISO-abbreviation] Invest New Drugs
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA095020
  • [Publication-type] Case Reports; Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Nitroso Compounds; 0 / nitrosylcobalamin; P6YC3EG204 / Vitamin B 12
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94. Coffin CM, Cassity J, Viskochil D, Randall RL, Albritton K: Non-neurogenic sarcomas in four children and young adults with neurofibromatosis type 1. Am J Med Genet A; 2004 May 15;127A(1):40-3
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  • [Title] Non-neurogenic sarcomas in four children and young adults with neurofibromatosis type 1.
  • It is well known that children and young adults with neurofibromatosis type 1 (NF1) have a higher risk for non-neurogenic sarcomas than the general population, in addition to an increased risk for malignant peripheral nerve sheath tumor.
  • When non-neurogenic sarcomas occur in early childhood, a subsequent malignant peripheral nerve sheath tumor can occur as a second malignant neoplasm, especially after alkylating agent chemotherapy and irradiation.
  • This report includes the clinicopathologic features of non-neurogenic sarcomas and secondary malignant peripheral nerve sheath tumor in the context of four cases of NF1.
  • The purpose is to emphasize that early diagnosis of NF1 and recognition of potential manifestations of non-neurogenic sarcomas are important for clinical care of these patients and their families.
  • [MeSH-major] Neoplasms, Second Primary / diagnosis. Neurofibromatosis 1 / diagnosis. Sarcoma / diagnosis
  • [MeSH-minor] Adolescent. Adult. Child, Preschool. Female. Humans. Male. Nerve Sheath Neoplasms / genetics. Nerve Sheath Neoplasms / secondary. Phyllodes Tumor / diagnosis. Precursor Cell Lymphoblastic Leukemia-Lymphoma / genetics. Prognosis. Rhabdomyosarcoma, Embryonal / diagnosis. Rhabdomyosarcoma, Embryonal / genetics


95. Rizvi S, Mehboob J, Asghar AH, Mateen A, Raza T, Hameed A: Omental caking: a rare manifestation of malignant peripheral nerve sheath tumour. J Coll Physicians Surg Pak; 2010 Aug;20(8):554-5
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  • [Title] Omental caking: a rare manifestation of malignant peripheral nerve sheath tumour.
  • Malignant peripheral nerve sheath tumour (MPNST) is a very rare tumour with an incidence of one per 100,000 and constitutes between 3 to 10% of all soft tissue sarcomas.
  • Mass responded well to chemotherapy comprising of Ifosfamide and Doxorubicin.
  • [MeSH-major] Nerve Sheath Neoplasms / pathology. Nerve Sheath Neoplasms / radiography. Omentum. Peritoneal Neoplasms / pathology. Peritoneal Neoplasms / radiography. Tomography, X-Ray Computed

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  • (PMID = 20688026.001).
  • [ISSN] 1022-386X
  • [Journal-full-title] Journal of the College of Physicians and Surgeons--Pakistan : JCPSP
  • [ISO-abbreviation] J Coll Physicians Surg Pak
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Pakistan
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96. Hoshimoto S, Morise Z, Takeura C, Ikeda M, Kagawa T, Tanahashi Y, Okabe Y, Mizoguchi Y, Sugioka A: Malignant Triton tumor in the retroperitoneal space associated with neurofibromatosis type 1: a case study. Rare Tumors; 2009;1(2):e27
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  • [Title] Malignant Triton tumor in the retroperitoneal space associated with neurofibromatosis type 1: a case study.
  • We report an extremely rare case of malignant Triton tumor developing in the retroperitoneal space in a patient with neurofibromatosis type 1.
  • A 21-year old man who had been diagnosed with neurofibromatosis type 1 was admitted to our hospital with the chief complaint of a palpable abdominal mass.
  • Abdominal computed tomography revealed a huge heterogeneous tumor measuring approximately 17 cm in diameter occupying the left retroperitoneal space, and numerous metastatic lesions between the left psoas muscle and the left thigh with dissolution of the left hip joint.
  • After the diagnosis of a retroperitoneal malignant neurogenic tumor, resection of the tumor with reconstruction of the abdominal aorta was conducted, followed by postoperative transarterial infusion chemotherapy.
  • The histopathological diagnosis was malignant peripheral nerve sheath tumor with rhabdomyosarcomatous differentiation, namely malignant Triton tumor.
  • Postoperative chemotherapy was in vain and the patient died 14 months after the surgery as a result of lung metastasis.

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  • (PMID = 21139906.001).
  • [ISSN] 2036-3613
  • [Journal-full-title] Rare tumors
  • [ISO-abbreviation] Rare Tumors
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Italy
  • [Other-IDs] NLM/ PMC2994481
  • [Keywords] NOTNLM ; malignant Triton tumor / malignant peripheral nerve sheath tumor / neurofibromatosis / retroperitoneal tumor.
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97. Nakayama J, Tanaka T, Furumura M, Takahashi A, Yamaguchi T, Shimura H, Ikeda S, Kuroki M: Inhibition of the proliferation of a malignant peripheral nerve sheath tumor cell line by gamma interferon gene transfection. J Dermatol; 2003 Dec;30(12):879-85
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  • [Title] Inhibition of the proliferation of a malignant peripheral nerve sheath tumor cell line by gamma interferon gene transfection.
  • A solitary malignant peripheral nerve sheath tumor was resected, and a tumor cell line was obtained using an explant culture.
  • The morphology of this cell line was quite similar to those of cell lines of dermal neurofibromas of neurofibromatosis 1, except for the fact that the malignant peripheral nerve sheath tumor cell line could be cultured for more than two months.
  • When the tumor cell line was transfected with human gamma interferon gene, the growth rate was remarkably abolished, as previously observed in neurofibroma cell lines transfected with the gamma interferon gene.
  • This new finding suggests the potential for treating either solitary or neurofibroma-related, inoperable, malignant peripheral nerve sheath tumors by local gamma interferon gene transfection in vivo.
  • [MeSH-major] Interferon-gamma / pharmacology. Neurofibromatosis 1 / therapy. Peripheral Nervous System Neoplasms / therapy. Transfection
  • [MeSH-minor] Aged. Cell Division / drug effects. Cell Division / genetics. Cell Line, Tumor / drug effects. Genetic Therapy. Humans. Male

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  • (PMID = 14739514.001).
  • [ISSN] 0385-2407
  • [Journal-full-title] The Journal of dermatology
  • [ISO-abbreviation] J. Dermatol.
  • [Language] eng
  • [Publication-type] Evaluation Studies; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Japan
  • [Chemical-registry-number] 82115-62-6 / Interferon-gamma
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98. Lehnhardt M, Daigeler A, Homann HH, Hauser J, Langer S, Steinsträsser L, Soimaru C, Puls A, Steinau HU: [Importance of specialized centers in diagnosis and treatment of extremity-soft tissue sarcomas. Review of 603 cases]. Chirurg; 2009 Apr;80(4):341-7
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  • [Title] [Importance of specialized centers in diagnosis and treatment of extremity-soft tissue sarcomas. Review of 603 cases].
  • [Transliterated title] Die Bedeutung von Referenzzentren in Diagnose und Therapie von Weichgewebssarkomen der Extremitäten. Auswertung von 603 Fällen.
  • Correct histopathologic diagnosis is essential for adequate treatment of soft tissue sarcomas.
  • Due to the disorder's rarity, multitude of subgroups, sometimes varying histopathologic appearance, and occasionally inadequate biopsy specimens, diagnosis and grading are challenging.
  • The records of 603 patients with soft tissue tumors of the extremities were reviewed concerning mismatches in primary and definite diagnoses relating to entity, evaluation of primary or recurrent tumor specimens, and the diagnosing pathology institution.
  • Liposarcoma and malignant fibrous histiocytoma were the most often diagnosed subgroups at 24% and 22.6%, respectively.
  • In the eight most frequent sarcoma types, malignant peripheral nerve sheath tumors and leiomyosarcoma had the highest rates of false primary diagnosis, 78.4% and 74.2% of cases, respectively.
  • For optimal treatment of soft tissue sarcomas, we suggest obtaining expert second opinion to ensure adequate surgical therapy and precise indications for radiation and chemotherapy.
  • [MeSH-major] Cancer Care Facilities. Extremities / surgery. Hospitals, Special. Hospitals, University. Sarcoma / diagnosis. Sarcoma / surgery. Soft Tissue Neoplasms / diagnosis. Soft Tissue Neoplasms / surgery
  • [MeSH-minor] Adolescent. Adult. Aged. Aged, 80 and over. Chemotherapy, Adjuvant. Child. Combined Modality Therapy. Diagnostic Errors. Female. Germany. Histiocytoma, Benign Fibrous / diagnosis. Histiocytoma, Benign Fibrous / pathology. Histiocytoma, Benign Fibrous / surgery. Humans. Leiomyosarcoma / diagnosis. Leiomyosarcoma / pathology. Leiomyosarcoma / surgery. Liposarcoma / diagnosis. Liposarcoma / pathology. Liposarcoma / surgery. Male. Middle Aged. Neoplasm Staging. Nerve Sheath Neoplasms / diagnosis. Nerve Sheath Neoplasms / pathology. Nerve Sheath Neoplasms / surgery. Radiotherapy, Adjuvant. Referral and Consultation. Retrospective Studies. Young Adult

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  • (PMID = 18523742.001).
  • [ISSN] 1433-0385
  • [Journal-full-title] Der Chirurg; Zeitschrift fur alle Gebiete der operativen Medizen
  • [ISO-abbreviation] Chirurg
  • [Language] ger
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Germany
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99. Pusceddu S, Bajetta E, Buzzoni R, Carcangiu ML, Platania M, Del Vecchio M, Ditto A: Primary uterine cervix melanoma resembling malignant peripheral nerve sheath tumor: a case report. Int J Gynecol Pathol; 2008 Oct;27(4):596-600
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  • [Title] Primary uterine cervix melanoma resembling malignant peripheral nerve sheath tumor: a case report.
  • A rare variant of malignant melanoma (MM) of the uterine cervix that mimics a malignant peripheral nerve sheath tumor (MPNST) is described.
  • Neoadjuvant chemotherapy and total abdominal hysterectomy and bilateral salpingo-ovariectomy plus pelvic lymphadenectomy were performed, and the diagnosis was MPNST, FIGO IIB.
  • A pathological review was obtained in our institution by a gynecological pathologist, who defined the primary neoplasm in the cervix as an MM, with a pattern of growth histologically simulating an MPNST, metastatic to the vagina.
  • To our knowledge, this is the first report in literature of MM of the uterine cervix resembling MPNST.
  • Despite its rarity, this variant of MM should be considered when a diagnosis of cervix MPNST is made.
  • The histological and immunohistochemical features of these different entities should be considered in the differential diagnosis.
  • [MeSH-major] Melanoma / pathology. Nerve Sheath Neoplasms / pathology. Uterine Cervical Neoplasms / pathology
  • [MeSH-minor] Adult. Diagnosis, Differential. Female. Histocytochemistry. Humans


100. Miller SJ, Rangwala F, Williams J, Ackerman P, Kong S, Jegga AG, Kaiser S, Aronow BJ, Frahm S, Kluwe L, Mautner V, Upadhyaya M, Muir D, Wallace M, Hagen J, Quelle DE, Watson MA, Perry A, Gutmann DH, Ratner N: Large-scale molecular comparison of human schwann cells to malignant peripheral nerve sheath tumor cell lines and tissues. Cancer Res; 2006 Mar 1;66(5):2584-91
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Large-scale molecular comparison of human schwann cells to malignant peripheral nerve sheath tumor cell lines and tissues.
  • Malignant peripheral nerve sheath tumors (MPNST) are highly invasive soft tissue sarcomas that arise within the peripheral nerve and frequently metastasize.
  • To identify molecular events contributing to malignant transformation in peripheral nerve, we compared eight cell lines derived from MPNSTs and seven normal human Schwann cell samples.
  • We found that MPNST lines are heterogeneous in their in vitro growth rates and exhibit diverse alterations in expression of pRb, p53, p14(Arf), and p16(INK4a) proteins.
  • All MPNST cell lines express the epidermal growth factor receptor and lack S100beta protein.
  • Global gene expression profiling using Affymetrix oligonucleotide microarrays identified a 159-gene molecular signature distinguishing MPNST cell lines from normal Schwann cells, which was validated in Affymetrix microarray data generated from 45 primary MPNSTs.
  • Expression of Schwann cell differentiation markers (SOX10, CNP, PMP22, and NGFR) was down-regulated in MPNSTs whereas neural crest stem cell markers, SOX9 and TWIST1, were overexpressed in MPNSTs.
  • Previous studies have implicated TWIST1 in apoptosis inhibition, resistance to chemotherapy, and metastasis.
  • Reducing TWIST1 expression in MPNST cells using small interfering RNA did not affect apoptosis or chemoresistance but inhibited cell chemotaxis.
  • Our results highlight the use of gene expression profiling in identifying genes and molecular pathways that are potential biomarkers and/or therapeutic targets for treatment of MPNST and support the use of the MPNST cell lines as a primary analytic tool.
  • [MeSH-major] Nerve Sheath Neoplasms / genetics. Schwann Cells / physiology
  • [MeSH-minor] Apoptosis / physiology. Cell Line, Tumor. Cell Movement / physiology. Drug Resistance, Neoplasm. Gene Expression Profiling. Humans. Nuclear Proteins / biosynthesis. Nuclear Proteins / genetics. Oligonucleotide Array Sequence Analysis. RNA, Small Interfering / genetics. Transfection. Twist Transcription Factor / biosynthesis. Twist Transcription Factor / genetics