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1. Furuno Y, Nishimura S, Kamiyama H, Numagami Y, Saito A, Kaimori M, Nishijima M: Intracranial peripheral-type primitive neuroectodermal tumor. Neurol Med Chir (Tokyo); 2008 Feb;48(2):72-6
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  • [Title] Intracranial peripheral-type primitive neuroectodermal tumor.
  • Magnetic resonance (MR) imaging revealed a large extraaxial tumor with cyst at the right frontotemporal region.
  • The solid part of the tumor was homogeneously enhanced on T(1)-weighted MR imaging after injection of gadolinium.
  • Digital subtraction angiography of the external carotid artery revealed sunburst appearance corresponding to the tumor, which was fed by the right middle meningeal artery.
  • His headache worsened and computed tomography revealed enlargement of the tumor and intracystic hemorrhage, so emergent operation was performed.
  • At surgery, the tumor strongly adhered to the dural membrane, and was obviously extraaxial.
  • The tumor and cyst were gross totally removed.
  • The histological diagnosis was peripheral-type primitive neuroectodermal tumor (pPNET).
  • Following surgery, radiation therapy and chemotherapy were given.
  • Ewing's sarcoma and pPNET form a family of small round cell tumors arising in the bone or soft tissue.
  • MIC-2 is a useful marker in the differential diagnosis.
  • [MeSH-major] Brain Neoplasms / pathology. Meningioma / pathology. Neuroectodermal Tumors, Primitive, Peripheral / pathology
  • [MeSH-minor] Adolescent. Antigens, CD / metabolism. Biomarkers, Tumor / metabolism. Cell Adhesion Molecules / metabolism. Diagnosis, Differential. Frontal Lobe / pathology. Headache / etiology. Headache / pathology. Humans. Ki-67 Antigen / metabolism. Magnetic Resonance Imaging. Male. Temporal Lobe / pathology. Tomography, X-Ray Computed

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  • (PMID = 18296876.001).
  • [ISSN] 0470-8105
  • [Journal-full-title] Neurologia medico-chirurgica
  • [ISO-abbreviation] Neurol. Med. Chir. (Tokyo)
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / Antigens, CD; 0 / Biomarkers, Tumor; 0 / CD99 protein, human; 0 / Cell Adhesion Molecules; 0 / Ki-67 Antigen
  • [Number-of-references] 20
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2. Lee YY, Kim DH, Lee JH, Choi JS, In KH, Oh YW, Cho KH, Roh YK: Primary pulmonary Ewing's sarcoma/primitive neuroectodermal tumor in a 67-year-old man. J Korean Med Sci; 2007 Sep;22 Suppl:S159-63
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  • [Title] Primary pulmonary Ewing's sarcoma/primitive neuroectodermal tumor in a 67-year-old man.
  • Extraskeletal Ewing's sarcoma (EES) is a branch of neuroectodermal tumor (PNET), which is very rare soft tissue sarcoma.
  • We report a case of EES/PNET arising is the lung of a 67-yr-old man.
  • Computed tomography, bone scintigraphy, and positron emission tomography confirmed the mass to have a primary pulmonary origin.
  • The diagnosis was confirmed both pathologically and genetically.
  • The mass lesion was resected, and the patient is currently undergoing chemotherapy.
  • [MeSH-major] Lung Neoplasms / diagnosis. Neuroectodermal Tumors, Primitive, Peripheral / diagnosis. Sarcoma, Ewing / diagnosis
  • [MeSH-minor] Aged. Calmodulin-Binding Proteins / genetics. Chromosome Breakage. Chromosomes, Human, Pair 22 / genetics. Diagnosis, Differential. Humans. Immunohistochemistry. In Situ Hybridization, Fluorescence. Male. RNA-Binding Proteins / genetics

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  • [Cites] Ann Thorac Surg. 2004 Aug;78(2):715-7 [15276562.001]
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  • (PMID = 17923745.001).
  • [ISSN] 1011-8934
  • [Journal-full-title] Journal of Korean medical science
  • [ISO-abbreviation] J. Korean Med. Sci.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Korea (South)
  • [Chemical-registry-number] 0 / Calmodulin-Binding Proteins; 0 / EWSR1 protein, human; 0 / RNA-Binding Proteins
  • [Other-IDs] NLM/ PMC2694395
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3. Suarez CR, Bertolone SJ, Raj AB, Coventry S: Second malignant neoplasms in childhood acute lymphoblastic leukemia: primitive neuroectodermal tumor of the chest wall with germline p53 mutation as a second malignant neoplasm. Am J Hematol; 2004 May;76(1):52-6
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  • [Title] Second malignant neoplasms in childhood acute lymphoblastic leukemia: primitive neuroectodermal tumor of the chest wall with germline p53 mutation as a second malignant neoplasm.
  • Second malignant neoplasm (SMNs) are a devastating sequelae observed on these children, with an estimated cumulative risk of 2-3.3% fifteen years after diagnosis.
  • Primitive neuroectodermal tumor of bone (PNET) is rarely observed as a SMN following treatment of childhood ALL.
  • The authors described the occurrence of a chest wall PNET of the bone at the site of a central line placement associated with both germ-line and tumor cell p53 mutation in a 8-year-old boy 1 year after completing therapy for standard risk ALL.
  • A review of the literature of 25,051 children treated for ALL discovered 230 SMNs (0.99%), and only one case of PNET of the bone was noted among this group.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Neoplasms, Second Primary / genetics. Neuroectodermal Tumors, Primitive, Peripheral / genetics. Precursor Cell Lymphoblastic Leukemia-Lymphoma / drug therapy. Thoracic Wall / pathology. Tumor Suppressor Protein p53 / genetics
  • [MeSH-minor] Child, Preschool. Combined Modality Therapy. Humans. Male. Mutation. Treatment Outcome

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  • [Copyright] Copyright 2004 Wiley-Liss, Inc.
  • (PMID = 15114597.001).
  • [ISSN] 0361-8609
  • [Journal-full-title] American journal of hematology
  • [ISO-abbreviation] Am. J. Hematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Tumor Suppressor Protein p53
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4. Laffosse JM, Accadbled F, Abid A, Kany J, Darodes P, Sales De Gauzy J: [Reconstruction of long bone defects with a vascularized fibular graft after tumor resection in children and adolescents: thirteen cases with 50-month follow-up]. Rev Chir Orthop Reparatrice Appar Mot; 2007 Oct;93(6):555-63
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  • [Title] [Reconstruction of long bone defects with a vascularized fibular graft after tumor resection in children and adolescents: thirteen cases with 50-month follow-up].
  • [Transliterated title] Reconstruction osseuse des os longs après exérèse carcinologique par l'utilisation de greffons fibulaires vascularisés chez l'enfant et l'adolescent.
  • PURPOSE OF THE STUDY: The vascularized fibular graft is a widely used technique for the reconstruction of long bone defects after tumor resection.
  • We report our experience with long bone reconstructions in children and adolescents after resection of primary malignant bone tumors.
  • Preoperatively, the pathological diagnosis was Ewing tumor (n=7), osteogenic sarcoma (n=5), neuroepithelioma (n=1).
  • All patients except one were given chemotherapy preoperatively and postoperatively and four received adjuvant radiotherapy.
  • Tumor resection created a gap (n=8) or involved resection-arthrodesis (n=5, three knees, one ankle, one elbow).
  • Eleven of the twelve patients who underwent tumor resection involving the lower limb were able to walk with full weight bearing at 13.9 months (range 841 months), half of them without any supportive device.
  • Among the fibular grafts which healed, primary healing of the distal end was noted in all cases, but not for the proximal end.
  • Healing was always achieved for the distal focus but not for the proximal focus which receives its blood supply from a branch of the anterior tibial artery which is not harvested.
  • The defective blood supply can thus hinder bone healing.
  • CONCLUSION: Long bone reconstruction using an autologous vascularized fibular graft is a reliable technique providing satisfactory functional results.
  • [MeSH-major] Bone Neoplasms / surgery. Bone Transplantation / methods. Reconstructive Surgical Procedures / methods. Surgical Flaps
  • [MeSH-minor] Adolescent. Arthrodesis / methods. Cause of Death. Chemotherapy, Adjuvant. Child. Child, Preschool. Follow-Up Studies. Fractures, Bone / etiology. Fractures, Spontaneous / etiology. Humans. Knee Joint / surgery. Lung Neoplasms / secondary. Neuroectodermal Tumors, Primitive, Peripheral / surgery. Osteosarcoma / surgery. Postoperative Complications. Radiotherapy, Adjuvant. Retrospective Studies. Sarcoma, Ewing / surgery. Treatment Outcome. Walking / physiology

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  • (PMID = 18065864.001).
  • [ISSN] 0035-1040
  • [Journal-full-title] Revue de chirurgie orthopédique et réparatrice de l'appareil moteur
  • [ISO-abbreviation] Rev Chir Orthop Reparatrice Appar Mot
  • [Language] fre
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] France
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5. Attabib NA, West M, Rhodes RH: Peripheral primitive neuroectodermal tumor of the cavernous sinus: case report. Neurosurgery; 2006 May;58(5):E992; discussion E992
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  • [Title] Peripheral primitive neuroectodermal tumor of the cavernous sinus: case report.
  • OBJECTIVE: Ewing sarcoma/peripheral primitive neuroectodermal tumors (pPNET family) are small, round, blue cell tumors that have a decided predilection for young patients and commonly arise in bone and soft tissue.
  • INTERVENTION: The patient underwent debulking of the tumor, and the diagnosis of a pPNET was made based on histological, immunohistochemical, and molecular genetics (EWS-FLI1 fusion gene) findings.
  • Bone scans, bone marrow aspiration, and biopsy and chest computed tomographic scans showed no evidence of systemic involvement.
  • The patient had adjuvant treatment with radiotherapy and chemotherapy.
  • After 14 months, the patient had no neurological deficits, and neuroimaging showed stable disease, although some chemotherapy complications occurred.
  • CONCLUSION: This is a case of cavernous sinus pPNET in a 48-year-old woman, in whom the diagnosis is supported by the presence of EWS-FLI1 fusion gene.
  • [MeSH-major] Cavernous Sinus / pathology. Neuroectodermal Tumors, Primitive, Peripheral / diagnosis. Neuroectodermal Tumors, Primitive, Peripheral / genetics

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  • (PMID = 16639307.001).
  • [ISSN] 1524-4040
  • [Journal-full-title] Neurosurgery
  • [ISO-abbreviation] Neurosurgery
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / EWS-FLI fusion protein; 0 / Oncogene Proteins, Fusion; 0 / Proto-Oncogene Protein c-fli-1; 0 / RNA-Binding Protein EWS
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6. Moschovi M, Trimis G, Stefanaki K, Anastasopoulos J, Syriopoulou V, Koultouki E, Tzortzatou-Stathopoulou F: Favorable outcome of Ewing sarcoma family tumors to multiagent intensive preoperative chemotherapy: a single institution experience. J Surg Oncol; 2005 Mar 15;89(4):239-43
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  • [Title] Favorable outcome of Ewing sarcoma family tumors to multiagent intensive preoperative chemotherapy: a single institution experience.
  • BACKGROUND: Aim of our study was to evaluate the efficacy of multiagent intensive preoperative chemotherapy in patients with Ewing sarcoma family tumors (ESFT), in order to succeed a better percentage of necrosis before surgical resection.
  • PROCEDURE: Eighteen patients with ESFT were treated with the same multiagent intensive preoperative protocol.
  • 5/18 patients had bone Ewings sarcoma (EWS) and 13/18 had peripheral primitive neuroectodermal tumor (PNET).
  • None had metastases at diagnosis.
  • Chemotherapy consisted of 5 or 6 cycles with vincristine, cisplatin, cyclophosphamide, and Adriamycin, followed by 12 cycles of vincristine, cyclophosphamide, and actinomycin-D.
  • Five patients with EWS underwent total resection after 5-6 cycles of preoperative chemotherapy and prosthetic replacement was performed in two of them.
  • In 3/13 patients with PNET the tumor was resected at diagnosis and in 1/13 after 5 cycles of chemotherapy, while 9/13 patients received chemotherapy only and/or radiotherapy.
  • RESULTS: In patients with EWS, the histologic specimens of the resected tumors showed that tissue necrosis was 100% in four patients and 95% in one patient.
  • No patient had topical recurrence or developed metastatic disease during follow-up period (2-13 years, mean time 7.4 years).
  • There were no major side effects of chemotherapy.
  • CONCLUSIONS: The intensive chemotherapy schedule, comprising of 5-6 cycles preoperatively, seems to maximize the percentage of tumor necrosis, thus improving outcome.
  • Our study implies that this combined therapy may improve the prognosis of ESFT.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Bone Neoplasms / drug therapy. Bone Neoplasms / genetics. Sarcoma, Ewing / drug therapy. Sarcoma, Ewing / genetics
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Cisplatin / administration & dosage. Combined Modality Therapy. Cyclophosphamide / administration & dosage. Doxorubicin / administration & dosage. Drug Administration Schedule. Female. Humans. Infant. Male. Neuroectodermal Tumors, Primitive, Peripheral / drug therapy. Neuroectodermal Tumors, Primitive, Peripheral / genetics. Neuroectodermal Tumors, Primitive, Peripheral / mortality. Neuroectodermal Tumors, Primitive, Peripheral / surgery. Preoperative Care. Radiotherapy Dosage. Survival Rate. Treatment Outcome. Vincristine / administration & dosage

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  • [Copyright] (c) 2005 Wiley-Liss, Inc.
  • (PMID = 15726621.001).
  • [ISSN] 0022-4790
  • [Journal-full-title] Journal of surgical oncology
  • [ISO-abbreviation] J Surg Oncol
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 5J49Q6B70F / Vincristine; 80168379AG / Doxorubicin; 8N3DW7272P / Cyclophosphamide; Q20Q21Q62J / Cisplatin
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7. Cebrián JL, Ibarzabal A, Garcia-Crespo R, Marco F, Ortega L, López-Durán L: Peripheral primitive neuroectodermal tumor after radiotherapy. Clin Orthop Relat Res; 2003 Aug;(413):255-60

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  • [Title] Peripheral primitive neuroectodermal tumor after radiotherapy.
  • A 41-year-old man had a peripheral neuroectodermal tumor develop at the distal third of the fibula 4 years after radiotherapy for relapsed villonodular synovitis.
  • This type of sarcoma usually is classified into the heterogeneic group of small round-cell bone tumors as a subdivision of Ewing's sarcomas.
  • The immuno-staining positivity of the neoplastic cells for the neuron-specific enolase allowed the authors to make the diagnosis of a tumor with neuroectodermal origin.
  • When the histologic study confirmed the diagnosis, the patient was treated with chemotherapy, surgical excision of the tumor, and adjuvant radiotherapy.
  • Radiotherapy is thought to be involved in the genesis of osteogenic sarcomas as it has been shown in several reports, but there is no evidence in the literature of a peripheral neuroectodermal tumor developing after radiotherapy.
  • [MeSH-major] Neoplasms, Second Primary / etiology. Neuroectodermal Tumors, Primitive, Peripheral / etiology. Synovitis, Pigmented Villonodular / radiotherapy

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  • (PMID = 12897617.001).
  • [ISSN] 0009-921X
  • [Journal-full-title] Clinical orthopaedics and related research
  • [ISO-abbreviation] Clin. Orthop. Relat. Res.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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8. Scurr M, Judson I: How to treat the Ewing's family of sarcomas in adult patients. Oncologist; 2006 Jan;11(1):65-72
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  • Ewing's sarcoma, peripheral primitive neuroectodermal tumor, and Askin's tumor comprise a single family of tumors, the Ewing's family of tumors, which is characterized by chromosomal translocation.
  • Ewing's sarcoma is known as a malignancy of childhood, but with a median age of 15 years at diagnosis, it should equally be regarded as a malignancy of adolescence and young adulthood.
  • There is much controversy regarding the role of age at diagnosis, with some studies showing older age to be associated with poorer outcome and others showing no association between age and survival.
  • This article examines whether age does affect outcome and treatment in this group of tumors.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Bone Neoplasms / drug therapy. Sarcoma, Ewing / drug therapy

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  • (PMID = 16401715.001).
  • [ISSN] 1083-7159
  • [Journal-full-title] The oncologist
  • [ISO-abbreviation] Oncologist
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 51
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9. Saada E, Thariat J, Follana P, Birtwisle-Peyrottes I, Haudebourg J, Trojani C, Bacque P, Thyss A: Primitive neuroectodermal tumor of the pelvis in an elderly patient. Onkologie; 2009 Sep;32(8-9):499-502
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  • [Title] Primitive neuroectodermal tumor of the pelvis in an elderly patient.
  • BACKGROUND: Peripheral primitive neuroectodermal tumors (PNET) belong to the rare family of primary bone neoplasms.
  • Recent clinicopathological studies have revealed that Ewing's sarcoma and PNET have overlapping features and they are now included in the same classification, the Ewing's sarcoma family of tumors (EFTs).
  • PNET have a marked predilection for the extremities and are very rare in the pelvis.
  • CASE REPORT: We report the case of a 69-year-old man with PNET sarcoma.
  • Outcome was favorable after combined modality treatment including chemotherapy based on the Memphis protocol - adapted from that used for Ewing's sarcoma in children - and surgery.
  • CONCLUSION: Our case is uncommon because of the age at diagnosis, the fortuitous way of revelation, and the choice of dose-intense chemotherapy adapted from the Memphis protocol (cyclophosphamide- and doxorubicin-based) for children, which was efficient and safe.
  • It supports the fact that an adult, and even an old patient, with good physical status, may be treated safely and radically even with dose-adapted aggressive chemotherapy.
  • [MeSH-major] Bone Neoplasms / pathology. Ilium / pathology. Neuroectodermal Tumors, Primitive / pathology. Pelvic Neoplasms / pathology

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  • (PMID = 19745594.001).
  • [ISSN] 1423-0240
  • [Journal-full-title] Onkologie
  • [ISO-abbreviation] Onkologie
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Switzerland
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10. Llombart-Bosch A, Pellín A, Carda C, Noguera R, Navarro S, Peydró-Olaya A: Soft tissue Ewing sarcoma--peripheral primitive neuroectodermal tumor with atypical clear cell pattern shows a new type of EWS-FEV fusion transcript. Diagn Mol Pathol; 2000 Sep;9(3):137-44
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  • [Title] Soft tissue Ewing sarcoma--peripheral primitive neuroectodermal tumor with atypical clear cell pattern shows a new type of EWS-FEV fusion transcript.
  • This study describes a new case of Ewing sarcoma (ES)-peripheral primitive neuroectodermal tumor (pPNET) with unusual phenotype and fusion gene structure.
  • The tumor located in the inguinal area of a 15-year-old boy showed a highly aggressive behavior with hematogenous metastases after intensive chemotherapy and bone marrow transplant, causing death 28 months after diagnosis.
  • The tumor displayed a clear cell pattern, and several neuroectodermal markers proved positive both in the original tumor and in xenografts.
  • This neuroectodermal character was confirmed by electron microscopy.
  • Moreover, cytogenetically the tumor has an unusual chromosomal rearrangement, t(2;22)(q13;q22,t(3;18)(p21;q23); representing a new EWS-FEV fusion type in which exon 7 of EWS gene is fused with exon 2 of FEV gene.
  • This is the third published study of an ES-pPNET showing EWS-FEV fusion described, but it is the first study of a tumor with the aforementioned fusion points.
  • [MeSH-major] Chromosomes, Human, Pair 2 / genetics. Chromosomes, Human, Pair 22 / genetics. Neuroectodermal Tumors, Primitive / genetics. Oncogene Proteins, Fusion / genetics. Sarcoma, Ewing / genetics. Soft Tissue Neoplasms / genetics. Translocation, Genetic / genetics
  • [MeSH-minor] Adolescent. Animals. Combined Modality Therapy. Disease Progression. Exons / genetics. Fatal Outcome. Groin. Humans. Karyotyping. Male. Mice. Mice, Nude. Neoplasm Metastasis. Neoplasm Proteins / analysis. Prognosis. Reverse Transcriptase Polymerase Chain Reaction. Transplantation, Heterologous. Tumor Cells, Cultured

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  • (PMID = 10976720.001).
  • [ISSN] 1052-9551
  • [Journal-full-title] Diagnostic molecular pathology : the American journal of surgical pathology, part B
  • [ISO-abbreviation] Diagn. Mol. Pathol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] UNITED STATES
  • [Chemical-registry-number] 0 / EWS-FEV fusion protein, human; 0 / Neoplasm Proteins; 0 / Oncogene Proteins, Fusion
  • [Number-of-references] 45
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11. Asano K, Kikuchi J, Munakata A, Ohkuma H, Kubo O: An infant case of intracranial peripheral-type primitive neuroectodermal tumor with long-term survival. Brain Tumor Pathol; 2007;24(2):69-74
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  • [Title] An infant case of intracranial peripheral-type primitive neuroectodermal tumor with long-term survival.
  • Supratentorial primitive neuroectodermal tumors (S-PNET) that develop in children have recently been classified into two types: central-type PNET (C-PNET), which has been reported over the years, and peripheral-type PNET (P-PNET), which develops intracranially and was referred to as Ewing's sarcoma in the past.
  • P-PNET is fundamentally a malignant tumor, but the patient reported here represents a case of long-term survival from onset without recurrence.
  • At the age of 21 months, a male infant developed a cranial bone deformity and symptoms of high intracranial pressure.
  • A CT scan revealed a cystic tumor attaching to the falx, and cyst drainage operation was immediately performed.
  • The intracranial tumor was then resected.
  • The tumor was an intradural extramedullary tumor, and it was totally excised with the falx attachment.
  • The tumor was initially diagnosed as a neuroblastoma, and postoperative treatment consisted of administration of radiotherapy and chemotherapy using cyclophosphamide and vincristine.
  • Recent repeated performance of histopathological analysis resulted in a diagnosis of P-PNET.
  • In recent years, studies in molecular biology have demonstrated that P-PNET involves the EWS-FLI1 chimeric gene, and immunohistochemical staining has shown P-PNET to be MIC2 positive.
  • P-PNET also differs from C-PNET with regard to prognosis, and for this reason it is believed that P-PNET and C-PNET should be considered separate entities.
  • That is, in spite of the fact that P-PNET is a malignant tumor, patient survival can be comparatively long.
  • Because P-PNET originates intracranially, it is fundamentally an intradural extramedullary tumor.
  • For this reason, treatment should consist of surgical excision that is as complete as possible, followed by appropriate radiotherapy and chemotherapy.
  • [MeSH-major] Diagnostic Errors. Neuroblastoma / pathology. Neuroectodermal Tumors, Primitive, Peripheral / pathology. Supratentorial Neoplasms / pathology
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Humans. Immunohistochemistry. Infant. Magnetic Resonance Imaging. Male. Neurosurgical Procedures. Radiotherapy. Tomography, X-Ray Computed

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  • (PMID = 18095134.001).
  • [ISSN] 1433-7398
  • [Journal-full-title] Brain tumor pathology
  • [ISO-abbreviation] Brain Tumor Pathol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Japan
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12. Kim KJ, Jang BW, Lee SK, Kim BK, Nam SL: A case of peripheral primitive neuroectodermal tumor of the ovary. Int J Gynecol Cancer; 2004 Mar-Apr;14(2):370-2
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  • [Title] A case of peripheral primitive neuroectodermal tumor of the ovary.
  • Peripheral primitive neuroectodermal tumor (PNET) belongs to the PNET/Ewing's sarcoma family.
  • PNET is a small round cell tumor of putative neuroectoderm origin and is the second most common sarcoma among children and young adults.
  • It may occur anywhere in the body and within any age group; however, it is most likely to occur in the bone and soft tissues.
  • There have been a small number of case reports of PNET arising in the ovary.
  • We presented a case of PNET arising in the right ovary of an 18-year-old woman.
  • The tumor was metastased to the lymph nodes of the pelvis and para-aorta at surgical staging.
  • We had persecuted Taxol/carboplatin chemotherapy, pelvic cavity radiotherapy, and Vincristine/Actinomycin, Cyclophosphamide/Doxorubicin (VACA).
  • [MeSH-major] Neuroectodermal Tumors / diagnosis. Ovarian Neoplasms / diagnosis
  • [MeSH-minor] Adolescent. Combined Modality Therapy. Diagnosis, Differential. Fatal Outcome. Female. Humans. Tomography, X-Ray Computed

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  • (PMID = 15086740.001).
  • [ISSN] 1048-891X
  • [Journal-full-title] International journal of gynecological cancer : official journal of the International Gynecological Cancer Society
  • [ISO-abbreviation] Int. J. Gynecol. Cancer
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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13. Heikaus S, Schaefer KL, Eucker J, Hogrebe E, Danebrock R, Wai DH, Krenn V, Gabbert HE, Poremba C: Primary peripheral primitive neuroectodermal tumor/Ewing's tumor of the testis in a 46-year-old man-differential diagnosis and review of the literature. Hum Pathol; 2009 Jun;40(6):893-7
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  • [Title] Primary peripheral primitive neuroectodermal tumor/Ewing's tumor of the testis in a 46-year-old man-differential diagnosis and review of the literature.
  • Peripheral primitive neuroectodermal tumor/Ewing's tumors are rare bone and soft tissue malignancies with a highly aggressive clinical course and early metastases occurring at multiple peripheral sites.
  • Here, we present for the first time a case of a 46-year-old man with a primary peripheral primitive neuroectodermal tumor/Ewing's tumor of the testis.
  • The diagnosis of peripheral primitive neuroectodermal tumor/Ewing's tumor was established by histology, immunohistochemistry, and molecular pathology.
  • The tumor revealed a rapid progress in 2 months' time.
  • Therefore, the patient was included in the EURO-E.W.I.N.G.99 study and was placed on chemotherapy.
  • However, the tumor progressed during ongoing therapy, and the patient died in March 2008.
  • In conclusion, though being reported here for the first time, peripheral primitive neuroectodermal tumor/Ewing's tumors should be considered in the differential diagnosis of blue round cell tumors of the testis.
  • A rapid and correct diagnosis of this entity is crucial for fast and accurate therapy, which is stressed by the fatal case presented here.
  • [MeSH-major] Neuroectodermal Tumors, Primitive, Peripheral / pathology. Sarcoma, Ewing / pathology. Testicular Neoplasms / pathology
  • [MeSH-minor] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Diagnosis, Differential. Fatal Outcome. Humans. Male. Middle Aged

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  • (PMID = 19269015.001).
  • [ISSN] 1532-8392
  • [Journal-full-title] Human pathology
  • [ISO-abbreviation] Hum. Pathol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] United States
  • [Number-of-references] 23
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14. Gunluoglu MZ, Kara HV, Demir A, Dincer SI: Results of multimodal treatment of two patients with thoracic primitive neuroectodermal tumor. Is surgery really helpful for survival? Thorac Cardiovasc Surg; 2007 Oct;55(7):460-1
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  • [Title] Results of multimodal treatment of two patients with thoracic primitive neuroectodermal tumor. Is surgery really helpful for survival?
  • Primitive neuroectodermal tumors (PNET) belong to the group of small round cell tumors and are rarely seen.
  • They are rapidly progressive tumors, despite usually being treated by a multimodal therapy which includes surgery and chemoradiotherapy.
  • We present two patients with PNET of the thorax treated in our clinic.
  • The first patient had a huge tumor in the right hemithorax, which shifted the mediastinum to the contralateral hemithorax.
  • Diagnosis was established by transthoracic fine-needle aspiration biopsy and the tumor was treated by surgical resection.
  • The second patient had a small tumor on the right costovertebral angle which protruded towards the skin and was diagnosed by incisional biopsy.
  • The tumor responded very well to preoperative chemotherapy and complete resection was achieved surgically.
  • This patient had bone metastasis, local recurrence and pleural pulmonary metastasis after 6, 18 and 28 months, respectively, and died 30 months after the operation.We discuss the limited effect of surgery on the treatment of thoracic PNET on the basis of the results of these patients in whom we performed surgery.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Bone Neoplasms / therapy. Brain Neoplasms / therapy. Neoplasm Recurrence, Local / therapy. Neuroectodermal Tumors, Primitive, Peripheral / therapy. Pleural Neoplasms / therapy. Thoracic Neoplasms / therapy. Thoracotomy
  • [MeSH-minor] Adult. Chemotherapy, Adjuvant. Combined Modality Therapy. Fatal Outcome. Female. Humans. Male. Neoadjuvant Therapy. Radiotherapy, Adjuvant. Tomography, X-Ray Computed

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  • (PMID = 17902071.001).
  • [ISSN] 0171-6425
  • [Journal-full-title] The Thoracic and cardiovascular surgeon
  • [ISO-abbreviation] Thorac Cardiovasc Surg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
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15. Utsunomiya A, Uenohara H, Suzuki S, Shimosaka S, Numagami Y, Nishimura S, Nishino A, Suzuki H, Sakurai Y: [A case of peripheral-type primitive neuroectodermal tumor arising in the dura mater at the frontal base]. No To Shinkei; 2004 Mar;56(3):237-41
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  • [Title] [A case of peripheral-type primitive neuroectodermal tumor arising in the dura mater at the frontal base].
  • Magnetic resonance image (MRI) revealed a large tumor in the frontal base with tumoral hemorrhage.
  • Angiography showed the tumor was fed by anterior meningeal arteries.
  • At surgery, the tumor was arising in the dura mater at the frontal base, and was removed totally.
  • Histological examination showed the tumor to be composed of small cells with uniform round nuclei and minimal cytoplasm.
  • The tumor was histologically confirmed to be peripheral-type primitive neuroectodermal tumor(pPNET).
  • Following surgery, he underwent whole brain, whole spine and local radiation therapy(30 Gy in total respectively) and received two 5-day cycles of chemotherapy, consisting of intravenous administration of cisplatin 20 mg/m2/day, etoposide 60 mg/m2/day and IFOS 900 mg/m2/day.
  • After these therapies, follow-up radiological examination showed there was no recurrence of the tumor for 24 months.
  • Ewing sarcoma and pPNET(ES/pPNET) is the designation given to a family of small round cell tumor arising in bone or soft tissues.
  • Intracranial PNETs are devided into central nervous system PNET(cPNET) and pPNET.
  • It is necessary that intracranial PNETs are divided into two types of PNETs because of different prognosis between these tumors.
  • MIC-2 is a specific marker for pPNET/ES family and is useful in the differential diagnosis of these two types of tumors.
  • [MeSH-major] Dura Mater. Meningeal Neoplasms / diagnosis. Neuroectodermal Tumors, Primitive, Peripheral / diagnosis
  • [MeSH-minor] Antigens, CD / analysis. Biomarkers, Tumor / analysis. Cell Adhesion Molecules / analysis. Chemotherapy, Adjuvant. Child. Diagnosis, Differential. Humans. Magnetic Resonance Angiography. Male. Radiotherapy, Adjuvant. Treatment Outcome

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  • (PMID = 15112448.001).
  • [ISSN] 0006-8969
  • [Journal-full-title] Nō to shinkei = Brain and nerve
  • [ISO-abbreviation] No To Shinkei
  • [Language] jpn
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / Antigens, CD; 0 / Biomarkers, Tumor; 0 / CD99 protein, human; 0 / Cell Adhesion Molecules
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16. Ludwig K: [Musculoskeletal lymphomas]. Radiologe; 2002 Dec;42(12):988-92
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  • Primary lymphomas of bone or skeletal muscle are rare entities.
  • The most frequent among these diseases are primary non-Hodgkin's lymphomas of bone.
  • They account for 3-5% of all bone tumors and 5% of all primary extranodal non-Hodgkin's lymphomas.
  • Primary manifestations of Hodgkin's disease in bone or skeletal muscle are rarities.
  • Primary non-Hodgkin's lymphomas of skeletal muscle are rarities as well.
  • Primary non-Hodgkin's lymphomas of bone can be found in any patient age.
  • The radiographic appearance of these entities resembles other aggressive bone tumors.
  • Their differential diagnosis includes -- depending on the patient's age -- Ewing's sarcoma,malignant fibrous histiocytoma,metastases of small cell tumors and osteomyelitis.Further differential diagnoses are the peripheral primitive neuroectodermal tumor (PNET), osteosarcoma, eosinophilic granuloma and fibrosarcoma.
  • Treatment of primary non-Hodgkin's lymphomas uses combinations of chemotherapy and radiation therapy.
  • Operative treatment is reserved for the treatment of complications.
  • The prognosis of primary non-Hodgkin's lymphomas is reflected by 10-year-survival-rates without recurrence of more than 80% in unifocal manifestations.
  • [MeSH-major] Bone Neoplasms / diagnosis. Hodgkin Disease / diagnosis. Lymphoma, Non-Hodgkin / diagnosis. Magnetic Resonance Imaging. Muscle Neoplasms / diagnosis. Tomography, X-Ray Computed
  • [MeSH-minor] Bone and Bones / pathology. Humans. Muscle, Skeletal / pathology. Neoplasm Staging. Prognosis. Sensitivity and Specificity

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  • (PMID = 12486552.001).
  • [ISSN] 0033-832X
  • [Journal-full-title] Der Radiologe
  • [ISO-abbreviation] Radiologe
  • [Language] ger
  • [Publication-type] English Abstract; Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 0
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17. Koscielniak E, Morgan M, Treuner J: Soft tissue sarcoma in children: prognosis and management. Paediatr Drugs; 2002;4(1):21-8
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  • [Title] Soft tissue sarcoma in children: prognosis and management.
  • Soft tissue sarcomas (STS) account for approximately 7% of malignant neoplasms in children.
  • The heterogeneity of STS makes the diagnosis and therapy particularly difficult and should be reserved for specialized centers with expertise in treating cancer in children.
  • Major progress in the accuracy of diagnosis and classification has been made by the identification of specific, recurring genetic alterations t(2;13)(q35;q14) and t(1;13)(p36;q14) in alveolar rhabdomyosarcomas (RMS), t(X;18)(p11;q11) for synovial sarcoma (SS) and t(11;22)(q24;q12) or t(21;22)(q22;q12) for Ewing's tumor family.
  • As a result of large multicenter STS studies, such as the North-American Intergroup Rhabdomyosarcoma Study, the German Pediatric Soft Tissue Sarcoma Study Group (CWS), Italian Gruppo Cooperativo Italiano study and Sociètè Internationale d'Oncologie Pèdiatrique (SIOP) Malignant Mesenchymal Tumors study, the identification of more effective treatment strategies and improvement in prognosis have been made in the last 30 years.
  • Prognostic variables were identified and, by exploring novel therapeutic strategies, criteria were established for the use of preoperative chemotherapy and radiotherapy, and primary and delayed second-look surgery.
  • As a result of evaluation of different drugs active in STS, refinements in the utilization of chemotherapy have been made possible.
  • Clinical trials have also been instrumental in defining the late effects of treatment.
  • In STS the following drugs have proven to be useful: dactinomycin, vincristine, alkylating agents such as cyclophosphamide and ifosfamide, as well as anthracyclines such as doxorubicin (adriamycin) and epi-doxorubicin.
  • Recommendations for radiation are dependent on the primary site and size of the tumour, histology, patient age and the extent of disease before and after surgical resection.
  • In general, with conventional fractionation (1 x 1.8 to 2 Gy/day) radiotherapy doses between 40 and 50 Gy should be administered.
  • The German CWS group explored the effectiveness of 32 Gy when accelerated and hyperfractionated, and given simultaneously to chemotherapy, and found it adequate for local tumor control in patients with selected favorable prognostic factors.
  • When treated with a combination of chemotherapy and local therapy, STS showed an event-free survival between 50 and 80% [RMS 70%, extraskeletal Ewing sarcoma (EES) and peripheral neuroectodermal tumor (PNET) circa 50%, and SS 70 to 80%].
  • Age (>10 years), bone, and/or bone marrow metastases are associated with a very poor prognosis (survival rate of about 5%).
  • The value of high dose chemotherapy with hematopoietic stem cell rescue in patients with poor prognostic STS remains unclear and should be performed in controlled studies only.
  • [MeSH-major] Sarcoma / diagnosis. Soft Tissue Neoplasms / diagnosis
  • [MeSH-minor] Antineoplastic Combined Chemotherapy Protocols. Child. Combined Modality Therapy. Humans. Neoplasm Staging. Prognosis. Radiotherapy. Rhabdomyosarcoma / diagnosis. Rhabdomyosarcoma / surgery. Rhabdomyosarcoma / therapy

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  • (PMID = 11817983.001).
  • [ISSN] 1174-5878
  • [Journal-full-title] Paediatric drugs
  • [ISO-abbreviation] Paediatr Drugs
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Switzerland
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18. Kano T, Sasaki A, Tomizawa S, Shibasaki T, Tamura M, Ohye C: Primary Ewing's sarcoma of the orbit: case report. Brain Tumor Pathol; 2009;26(2):95-100
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  • Computed tomography revealed a left intraorbital mass measuring 3 cm x 3 cm involving the left lateral wall of the orbit and the greater wing of the left sphenoid bone.
  • During surgery, the tumor was seen to arise from the lateral wall of the orbit and infiltrate into the left temporal muscle.
  • Following the surgery, the patient was administered radiation therapy for the whole cranium and chemotherapy for the residual tumors.
  • However, the tumor recurred, and the patient died about 2 years following the first surgery because the tumor had metastasized to the lung.
  • On light microscopy, the tumor cells were closely packed with uniform, small, and round cells.
  • Immunohistochemical studies showed that the tumor cell membrane stained positive for MIC2.
  • Based on these results, the tumor was diagnosed to be primary Ewing's sarcoma.
  • [MeSH-major] Neuroectodermal Tumors, Primitive, Peripheral / diagnosis. Orbital Neoplasms / diagnosis. Sarcoma, Ewing / diagnosis

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  • (PMID = 19856222.001).
  • [ISSN] 1861-387X
  • [Journal-full-title] Brain tumor pathology
  • [ISO-abbreviation] Brain Tumor Pathol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] TDQ283MPCW / Eosine Yellowish-(YS); YKM8PY2Z55 / Hematoxylin
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19. Sato S, Mitsuyama T, Ishii A, Kawakami M, Kawamata T: Multiple primary cranial Ewing's sarcoma in adulthood: case report. Neurosurgery; 2009 Feb;64(2):E384-6; discussion E386
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  • OBJECTIVE: Ewing's sarcoma is a malignant bone tumor occurring most frequently in the long bones and flat bones as a solitary lesion during the first 2 decades of life.
  • Ewing's sarcoma and peripheral primitive neuroectodermal tumor have recently been considered to be the same entity because of histological and molecular similarities.
  • A computed tomographic scan revealed osteolytic changes of the inner calvarial bone.
  • Electron microscopy showed little differentiation to neuronal tissue, indicating Ewing's sarcoma.
  • After surgical treatment, conventional whole cranial irradiation of 40 Gy and chemotherapy were conducted.
  • The tumor in the left frontal region disappeared.
  • CONCLUSION: Although quite rare, Ewing's sarcoma should be taken into consideration as a differential diagnosis of multiple cranial mass lesions in adulthood.
  • [MeSH-major] Neoplasms, Multiple Primary / diagnosis. Neoplasms, Multiple Primary / therapy. Sarcoma, Ewing / diagnosis. Sarcoma, Ewing / therapy. Skull Neoplasms / diagnosis. Skull Neoplasms / therapy
  • [MeSH-minor] Adult. Diagnosis, Differential. Humans. Male. Treatment Outcome

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  • (PMID = 19190443.001).
  • [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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20. Deutsch M, Wollman MR: Radiotherapy for metastases to the mandible in children. J Oral Maxillofac Surg; 2002 Mar;60(3):269-71
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  • Six children had a neuroblastoma, 1 had angiosarcoma of the liver, 1 had adenocarcinoma of the rectum, and 1 had peripheral primitive neuroectodermal tumor (Ewing's sarcoma) of the spine.
  • In 3 children, the mandible was the first bone involved by metastases.
  • All children had received chemotherapy.
  • RESULTS: All children died of disseminated disease at 5 to 59 months from their initial diagnosis, 5 to 29 months from the detection of metastases to bone, and only 6 days to 17 months (median, 2 months) from the first treatment of metastases to the mandible.
  • CONCLUSIONS: The outlook for children with metastases that involve the mandible is very poor, and we recommend short intensive courses of radiotherapy consisting of 1 to 3 treatments to total doses of 400 to 1,200 cGy for palliation of pain.

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  • [Copyright] Copyright 2002 American Association of Oral and Maxillofacial Surgeons
  • (PMID = 11887137.001).
  • [ISSN] 0278-2391
  • [Journal-full-title] Journal of oral and maxillofacial surgery : official journal of the American Association of Oral and Maxillofacial Surgeons
  • [ISO-abbreviation] J. Oral Maxillofac. Surg.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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21. de Alava E, Antonescu CR, Panizo A, Leung D, Meyers PA, Huvos AG, Pardo-Mindán FJ, Healey JH, Ladanyi M: Prognostic impact of P53 status in Ewing sarcoma. Cancer; 2000 Aug 15;89(4):783-92
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  • BACKGROUND: Disease stage at the time of diagnosis and response to therapy are the main prognostic factors for patients with Ewing sarcoma or peripheral neuroectodermal tumor (ES/PNET).
  • The primary genetic alteration in ES/PNET, the fusion of the EWS gene with FLI1 or ERG, is diagnostically highly specific for these tumors, and molecular variation in the structure of the EWS-FLI1 fusion gene also is of prognostic significance.
  • In contrast, secondary genetic alterations, such as P53 alterations, are relatively uncommon in ES/PNET, and their prognostic impact has not been extensively studied.
  • METHODS: Prechemotherapy, paraffin embedded, nondecalcified, primary tumor material in a well-characterized series of 55 patients with ES/PNET with defined EWS-FLI1 fusion transcripts (32 patients with type 1 and 23 patients with other types) was studied retrospectively by immunohistochemical techniques for cell cycle regulators and proliferative markers, such as P53, P21(WAF1), and Ki-67, as well as by the terminal deoxynucleotidyl transferase-mediated dUTP nick-end labeling (TUNEL) technique for apoptosis.
  • Nuclear P53 expression in > 20% of tumor cells was scored as aberrant overexpression.
  • Histologic response to neoadjuvant chemotherapy was assessed.
  • RESULTS: Aberrant P53 expression (in > 20% of tumor cells) was present in 6 patients (11%) but showed no statistically significant correlation with disease stage, tumor size, proliferation rate (Ki-67), apoptotic rate (TUNEL), or EWS-FLI1 fusion type.
  • In multivariate Cox analyses of overall survival, P53 > 20% was the strongest negative factor among prognostic factors available at the time of diagnosis (P = 0.001; relative risk [RR] = 9) and when chemotherapy response was included in the analysis (P53 > 20%: P = 0.01; RR = 10).
  • CONCLUSIONS: P53 alteration appears to define a small clinical subset of patients with ES/PNET with a markedly poor outcome.
  • [MeSH-major] Biomarkers, Tumor / metabolism. Bone Neoplasms / metabolism. Sarcoma, Ewing / metabolism. Tumor Suppressor Protein p53 / metabolism
  • [MeSH-minor] Adolescent. Adult. Antineoplastic Agents / therapeutic use. Child. Child, Preschool. Female. Humans. Immunohistochemistry. Male. Neoplasm Staging. Oncogene Proteins, Fusion / genetics. Oncogene Proteins, Fusion / metabolism. Point Mutation. Prognosis. Prospective Studies. Proto-Oncogene Protein c-fli-1. RNA-Binding Protein EWS. Transcription Factors / genetics. Transcription Factors / metabolism. Treatment Outcome

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  • [Copyright] Copyright 2000 American Cancer Society.
  • (PMID = 10951341.001).
  • [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
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Biomarkers, Tumor; 0 / EWS-FLI fusion protein; 0 / Oncogene Proteins, Fusion; 0 / Proto-Oncogene Protein c-fli-1; 0 / RNA-Binding Protein EWS; 0 / Transcription Factors; 0 / Tumor Suppressor Protein p53
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22. Lowichik A, Zhou H, Pysher TJ, Smith L, Lemons R, Coffin CM: Therapy associated changes in childhood tumors. Adv Anat Pathol; 2000 Nov;7(6):341-59
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  • [Title] Therapy associated changes in childhood tumors.
  • Contemporary treatment regimens for the common solid tumors of childhood have led to increased numbers of post-treatment pathologic specimens from survivors.
  • Current therapeutic strategies for childhood cancers in North America require an accurate pathologic diagnosis and stratify patients based on combinations of clinical, biological, and pathologic features.
  • In several tumor systems, the pathologic response to therapy also modifies the treatment regimen.
  • Accurate pathologic interpretation of such specimens is critical in providing useful prognostic information for therapeutic decisions.
  • Standardized handling of post-therapy pathologic specimens, appropriate use of molecular and genetic studies, consideration of the differential diagnoses, and assessment of the potential biologic significance of therapy-induced pathologic changes are, therefore, critical for patient management and determination of treatment protocols.
  • [MeSH-minor] Adolescent. Bone Neoplasms / drug therapy. Bone Neoplasms / pathology. Bone Neoplasms / surgery. Child. Child, Preschool. Fibrosarcoma / pathology. Fibrosarcoma / therapy. Hepatoblastoma / pathology. Hepatoblastoma / therapy. Histocytochemistry. Humans. Kidney Neoplasms / drug therapy. Kidney Neoplasms / pathology. Kidney Neoplasms / surgery. Liver Neoplasms / pathology. Liver Neoplasms / therapy. Neuroectodermal Tumors, Primitive, Peripheral / drug therapy. Neuroectodermal Tumors, Primitive, Peripheral / pathology. Osteosarcoma / pathology. Osteosarcoma / surgery. Prognosis. Rhabdomyosarcoma / drug therapy. Rhabdomyosarcoma / pathology. Rhabdomyosarcoma / surgery. Sarcoma, Ewing / drug therapy. Sarcoma, Ewing / pathology. Soft Tissue Neoplasms / drug therapy. Soft Tissue Neoplasms / pathology. Soft Tissue Neoplasms / surgery. Wilms Tumor / drug therapy. Wilms Tumor / pathology. Wilms Tumor / surgery

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  • (PMID = 11078058.001).
  • [ISSN] 1072-4109
  • [Journal-full-title] Advances in anatomic pathology
  • [ISO-abbreviation] Adv Anat Pathol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] UNITED STATES
  • [Number-of-references] 149
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