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1. Butturini AM, Jacob M, Aguajo J, Vander-Walde NA, Villablanca J, Jubran R, Erdreich-Epstein A, Marachelian A, Dhall G, Finlay JL: High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors: the impact of prior radiotherapy on outcome. Cancer; 2009 Jul 1;115(13):2956-63
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  • [Title] High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors: the impact of prior radiotherapy on outcome.
  • BACKGROUND: The role of myeloablative chemotherapy in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors (MB/ST-PNET) is controversial, in particular in patients who develop recurrent disease after craniospinal radiotherapy.
  • METHODS: In this retrospective analysis, the authors investigated the outcome of children with recurrent MB/ST-PNET who were referred for myeloablative chemotherapy and autologous hematopoietic progenitor cell rescue at Childrens Hospital Los Angeles.
  • RESULTS: Thirty-three children were referred for myeloablative chemotherapy: Fourteen of those children were never transplanted because of pre-transplant adverse events, and 19, including 6 without and 13 with previous irradiation, underwent transplant.
  • Nine previously irradiated children experienced 4 toxic deaths and 6 tumor recurrences (1 patient had both): An interval of <1 year between initial radiotherapy and myeloablative chemotherapy predicted a greater risk of toxic death (P = .02), whereas a history of meningeal metastases at diagnosis and a poor response to the initial rescue therapy predicted a greater risk of post-transplant recurrence (P = .03 and P = .08, respectively).
  • CONCLUSIONS: Myeloablative doses of thiotepa-based chemotherapy and radiotherapy were able to cure most children who had radiotherapy-naive, chemoresponsive recurrences.
  • Children who developed recurrences after craniospinal radiotherapy had poorer outcomes; however, cure was possible in those who had good prognostic features at presentation, chemoresponsive recurrences, and a long interval between initial radiotherapy and myeloablative chemotherapy.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Cranial Irradiation. Hematopoietic Stem Cell Transplantation. Medulloblastoma / therapy. Neuroectodermal Tumors, Primitive / therapy. Supratentorial Neoplasms / therapy. Thiotepa / administration & dosage
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Combined Modality Therapy. Disease-Free Survival. Female. Humans. Infant. Male. Neoplasm Recurrence, Local. Survival Rate

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  • (PMID = 19402050.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 905Z5W3GKH / Thiotepa
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2. Bisogno G, Riccardi R, Ruggiero A, Arcamone G, Prete A, Surico G, Provenzi M, Bertolini P, Paolucci P, Carli M: Phase II study of a protracted irinotecan schedule in children with refractory or recurrent soft tissue sarcoma. Cancer; 2006 Feb 1;106(3):703-7
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  • [Title] Phase II study of a protracted irinotecan schedule in children with refractory or recurrent soft tissue sarcoma.
  • The Italian Soft Tissue Sarcoma (STS) Committee performed a multiinstitutional Phase II study to evaluate its effect on STS.
  • Thirty patients, 13 with peripheral primitive neuroectodermal tumor (PNET), 12 with rhabdomyosarcoma (RMS), 3 with desmoplastic small round cell tumor (DSRCT), and 2 with other STS were evaluable for response.
  • The overall response rate was 23% (2 complete remissions +5 partial remissions of 30 patients), 38% for PNET and 16% for RMS.
  • CONCLUSIONS: As a single agent in the treatment of recurrent and refractory STS, irinotecan administered on a daily x5 x2 schedule revealed a noteworthy response rate in a population of heavily pretreated patients, especially in the subset of patients with PNET.
  • [MeSH-major] Antineoplastic Agents, Phytogenic / administration & dosage. Camptothecin / analogs & derivatives. Sarcoma / drug therapy. Soft Tissue Neoplasms / drug therapy
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Disease Progression. Drug Administration Schedule. Female. Humans. Infant. Infusions, Intravenous. Male. Neutropenia / chemically induced. Treatment Outcome

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  • [Copyright] Copyright (c) 2005 American Cancer Society.
  • (PMID = 16369989.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Clinical Trial, Phase II; Journal Article; Multicenter Study; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Phytogenic; 7673326042 / irinotecan; XT3Z54Z28A / Camptothecin
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3. Pérez Martínez A, Contra T, Scaglione C, Díaz Pérez MA, Madero López L: [Topotecan for pediatric patients with resistant and recurrent solid tumors]. An Pediatr (Barc); 2003 Aug;59(2):143-8
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  • [Title] [Topotecan for pediatric patients with resistant and recurrent solid tumors].
  • [Transliterated title] Topotecán en el tratamiento de niños con tumores sólidos refractarios o recidivantes.
  • BACKGROUND: Topotecan is a cytotoxic drug isolated from the Camptotheca acuminata tree (from China).
  • It is able to block the enzyme DNA topoisomerase I and has recently been used in the treatment of pediatric cancer.
  • OBJECTIVES: To evaluate our preliminary experience with topotecan in the second line treatment of refractory solid tumors in the pediatric age group.
  • PATIENTS AND MEHTODS: We performed a retrospective study of 10 patients with various recurrent solid tumors resistant to first line treatment who were treated with topotecan alone or in association with other chemotherapeutic agents.
  • RESULTS: Ten patients with recurrent solid tumors or tumors that were refractory to conventional treatment (two neuroblastomas, three rhabdomyosarcoma, two PNET/Ewing's sarcoma, one anaplastic astrocytoma, one soft tissue sarcoma and one synovial sarcoma) were included.
  • CONCLUSIONS: In our experience, topotecan is beneficial in some refractory or recurrent solid tumors, especially neuroblastomas and soft tissue sarcomas.
  • Patients with a complete response to topotecan could benefit from high-dose chemotherapy and autologous stem cell rescue therapy.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Neoplasms / drug therapy. Topotecan / therapeutic use
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Drug Administration Schedule. Drug Resistance, Neoplasm. Female. Humans. Infant. Male. Neoplasm Staging. Remission Induction. Retrospective Studies

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  • (PMID = 12882743.001).
  • [ISSN] 1695-4033
  • [Journal-full-title] Anales de pediatría (Barcelona, Spain : 2003)
  • [ISO-abbreviation] An Pediatr (Barc)
  • [Language] spa
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Spain
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 7M7YKX2N15 / Topotecan
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4. Kanamori M, Kumabe T, Saito R, Yamashita Y, Sonoda Y, Tominaga T: [The safety of combination chemotherapy with ifosfamide, cisplatin, and etoposide (ICE): single-institution retrospective review of 108 cases]. No Shinkei Geka; 2010 Nov;38(11):997-1005
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  • [Title] [The safety of combination chemotherapy with ifosfamide, cisplatin, and etoposide (ICE): single-institution retrospective review of 108 cases].
  • PURPOSE: The adverse effects of combination chemotherapy of ifosfamide, cisplatin, and etoposide (ICE) were evaluated in the treatment of various intracranial brain tumors.
  • The histological diagnosis was newly diagnosed or recurrent germ cell tumor in 45 cases, medulloblastoma in 19, primitive neuroectodermal tumor (PNET) in 7, anaplastic ependymoma in 6, recurrent glioblastoma in 13, and others in 18 cases.
  • Patients received 1-8 cycles of ICE chemotherapy with or without radiation therapy.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / adverse effects. Brain Neoplasms / drug therapy
  • [MeSH-minor] Adolescent. Adult. Antineoplastic Agents / administration & dosage. Antineoplastic Agents / adverse effects. Antineoplastic Agents, Alkylating / administration & dosage. Antineoplastic Agents, Alkylating / adverse effects. Antineoplastic Agents, Phytogenic / administration & dosage. Antineoplastic Agents, Phytogenic / adverse effects. Child. Child, Preschool. Cisplatin / administration & dosage. Cisplatin / adverse effects. Ependymoma / drug therapy. Etoposide / administration & dosage. Etoposide / adverse effects. Female. Glioblastoma / drug therapy. Hematologic Diseases / chemically induced. Humans. Ifosfamide / administration & dosage. Ifosfamide / adverse effects. Infant. Male. Medulloblastoma / drug therapy. Middle Aged. Neoplasms, Germ Cell and Embryonal / drug therapy. Neuroectodermal Tumors, Primitive / drug therapy. Retrospective Studies

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  • (PMID = 21081811.001).
  • [ISSN] 0301-2603
  • [Journal-full-title] No shinkei geka. Neurological surgery
  • [ISO-abbreviation] No Shinkei Geka
  • [Language] jpn
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Antineoplastic Agents, Alkylating; 0 / Antineoplastic Agents, Phytogenic; 6PLQ3CP4P3 / Etoposide; Q20Q21Q62J / Cisplatin; UM20QQM95Y / Ifosfamide
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5. Huang KH, Shun CT, Huang SY, Yu HJ, Chueh SC, Chen J: Primary primitive neuroectodermal tumor of the urinary tract. J Formos Med Assoc; 2006 Dec;105(12):1008-12

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  • [Title] Primary primitive neuroectodermal tumor of the urinary tract.
  • Primary primitive neuroectodermal tumor (PNET) of the urinary tract is a rare disease with aggressive behavior and poor prognosis.
  • Only three (0.035%) cases with PNET of the urinary tract were identified.
  • The first case was a 44-year-old man with left renal PNET who underwent hand-assisted laparoscopic radical nephrectomy and adjuvant chemotherapy.
  • There was no recurrent tumor at the 4-year follow-up.
  • The second case was a 75-year-old woman with right renal PNET with inferior vena cava (IVC) thrombosis extending to the right atrium.
  • The third case was a 45-year-old man with left ureteral PNET.
  • Tumor recurrence was noted 7 years later.
  • Urinary tract PNET appears to be an aggressive malignancy.

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  • (PMID = 17185243.001).
  • [ISSN] 0929-6646
  • [Journal-full-title] Journal of the Formosan Medical Association = Taiwan yi zhi
  • [ISO-abbreviation] J. Formos. Med. Assoc.
  • [Language] ENG
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Singapore
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6. Wolff JE, Finlay JL: High-dose chemotherapy in childhood brain tumors. Onkologie; 2004 Jun;27(3):239-45
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  • [Title] High-dose chemotherapy in childhood brain tumors.
  • Early attempts to use high-dose chemotherapy technology in order to improve the effect of nitrosourea on high-grade gliomas resulted in minimal benefit as well as in severe toxicity.
  • Since then, other drugs have been applied in conjunction with either autologous bone marrow or peripheral blood stem cells, including thiotepa, etoposide, melphalan, cyclophosphamide, and busulfan.
  • The data suggest benefit in recurrent primitive neuroectodermal tumors (PNET), in newly diagnosed young children with PNET and possibly in young children with newly diagnosed ependymoma, as a strategy not only to improve tumor-free survival but also to avoid exposure of the young brain to irradiation.
  • In other tumors such as recurrent ependymoma and newly diagnosed or recurrent brain stem glioma, high-dose chemotherapy remains ineffective.
  • New protocols under evaluation include new agents, multiple cycles of high-dose chemotherapy and allogeneic transplantation as immunotherapeutic approach.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Brain Neoplasms / drug therapy. Drug Therapy / methods
  • [MeSH-minor] Child. Child, Preschool. Combined Modality Therapy / methods. Ependymoma / drug therapy. Ependymoma / surgery. Glioma / drug therapy. Glioma / surgery. Humans. Infant. Infant, Newborn. Medulloblastoma / drug therapy. Medulloblastoma / surgery. Neuroectodermal Tumors / drug therapy. Neuroectodermal Tumors / surgery. Practice Patterns, Physicians'

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  • [Copyright] Copyright 2004 S. Karger GmbH, Freiburg
  • (PMID = 15249712.001).
  • [ISSN] 0378-584X
  • [Journal-full-title] Onkologie
  • [ISO-abbreviation] Onkologie
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Antineoplastic Agents
  • [Number-of-references] 65
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7. Nicholson HS, Kretschmar CS, Krailo M, Bernstein M, Kadota R, Fort D, Friedman H, Harris MB, Tedeschi-Blok N, Mazewski C, Sato J, Reaman GH: Phase 2 study of temozolomide in children and adolescents with recurrent central nervous system tumors: a report from the Children's Oncology Group. Cancer; 2007 Oct 1;110(7):1542-50
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  • [Title] Phase 2 study of temozolomide in children and adolescents with recurrent central nervous system tumors: a report from the Children's Oncology Group.
  • BACKGROUND: Effective chemotherapy is lacking for most types of central nervous system (CNS) tumors in children.
  • Temozolomide, an agent with activity against adult brain tumors, was investigated in children and adolescents with recurrent CNS tumors.
  • RESULTS: The cohort comprised 122 patients, including 113 with CNS tumors.
  • Among 104 evaluable patients with CNS tumors, 5 PRs and 1 CR were observed.
  • PRs occurred in 1 of 23 evaluable patients with high-grade astrocytoma, 1 of 21 with low-grade astrocytoma, and 3 of 25 with medulloblastoma/primitive neuroectodermal tumor (PNET).
  • The CR occurred in an additional patient with medulloblastoma/PNET.
  • No responses were observed in patients with ependymoma, brain-stem glioma, or other CNS tumors.
  • CONCLUSIONS: Although overall objective responses were limited, further exploration of temozolomide may be warranted in children with medulloblastoma and other PNETs, or in patients with low-grade astrocytoma, perhaps in a setting of less pretreatment than the patients in the current study, or in the context of multiagent therapy.
  • [MeSH-major] Antineoplastic Agents, Alkylating / therapeutic use. Brain Neoplasms / drug therapy. Dacarbazine / analogs & derivatives. Neoplasm Recurrence, Local / drug therapy
  • [MeSH-minor] Administration, Oral. Adolescent. Adult. Astrocytoma / drug therapy. Central Nervous System Neoplasms / drug therapy. Child. Child, Preschool. Drug Administration Schedule. Ependymoma / drug therapy. Female. Humans. Infant. Male. Medulloblastoma / drug therapy. Neuroectodermal Tumors, Primitive / drug therapy. Treatment Outcome


8. Fernandez MC, Krailo MD, Gerbing RR, Matthay KK: A phase I dose escalation of combination chemotherapy with granulocyte-macrophage-colony stimulating factor in patients with neuroblastoma. Cancer; 2000 Jun 15;88(12):2838-44
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  • [Title] A phase I dose escalation of combination chemotherapy with granulocyte-macrophage-colony stimulating factor in patients with neuroblastoma.
  • BACKGROUND: Dose intensity is important in the response to chemotherapy in patients with advanced neuroblastoma.
  • The aim of the current study was to determine the maximum tolerated dose of a combination chemotherapy regimen in the treatment of patients with recurrent neuroblastoma and peripheral neuroepithelioma (primitive neuroectodermal tumor [PNET]) and whether the use of growth factor would allow increased dose intensity.
  • METHODS: Twenty-nine patients diagnosed with recurrent neuroblastoma or PNET were treated with a combination chemotherapy regimen of cisplatin, 160 mg/m(2)/96 hours; doxorubicin, 40 mg/m(2)/96 hours; and escalated doses of etoposide and ifosfamide.
  • Granulocyte-macrophage-colony stimulating factor (GM-CSF) was administered beginning 24 hours after the completion of the chemotherapy.
  • A total of 12 of 29 patients developed a dose-limiting toxicity (DLT) after the first course of therapy.
  • Twenty-seven patients developed standard National Cancer Institute criteria Grade 3 or 4 toxicity after the first course of treatment and 7 patients achieved a complete or partial response to the first course.
  • CONCLUSIONS: This chemotherapy combination achieved a 31% overall response rate.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Granulocyte-Macrophage Colony-Stimulating Factor / therapeutic use. Neuroblastoma / drug therapy
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Cisplatin / administration & dosage. Dose-Response Relationship, Drug. Doxorubicin / administration & dosage. Drug Administration Schedule. Etoposide / administration & dosage. Female. Humans. Ifosfamide / administration & dosage. Infant. Infant, Newborn. Male. Neutropenia / chemically induced. Neutropenia / prevention & control. Thrombocytopenia / chemically induced. Thrombocytopenia / prevention & control. Treatment Outcome

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  • [Copyright] Copyright 2000 American Cancer Society.
  • (PMID = 10870069.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA 02971; United States / NCI NIH HHS / CA / CA 20320; United States / NCI NIH HHS / CA / CA13539; etc
  • [Publication-type] Clinical Trial; Clinical Trial, Phase I; Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] UNITED STATES
  • [Chemical-registry-number] 6PLQ3CP4P3 / Etoposide; 80168379AG / Doxorubicin; 83869-56-1 / Granulocyte-Macrophage Colony-Stimulating Factor; Q20Q21Q62J / Cisplatin; UM20QQM95Y / Ifosfamide; PAVI protocol
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9. Yoshida Y, Toma Y, Arai M, Higashi R, Kashihara K, Kaizaki Y: [Primitive neuroectodermal tumor arising 8 years after chemotherapy and radiotherapy for acute lymphoblastic leukemia: case report]. No Shinkei Geka; 2005 Jul;33(7):717-22
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  • [Title] [Primitive neuroectodermal tumor arising 8 years after chemotherapy and radiotherapy for acute lymphoblastic leukemia: case report].
  • We report a case of primitive neuroectodermal tumor (PNET) arising 8 years after chemotherapy and radiotherapy for acute lymphoblastic leukemia.
  • A 15-year-old boy with a history of acute lymphoblastic leukemia, at the age of 7, underwent chemotherapy and 14Gy of radiotherapy to the whole brain.
  • A gross total removal of the tumor was performed and histological examination showed it to be PNET.
  • Follow-up MRI showed no evidence of recurrent tumor 4 months after the radiotherapy.
  • This tumor was thought to be a secondary brain tumor arising in this survivor of childhood acute lymphoblastic leukemia and it is a rare complication of successful leukemia treatment.
  • [MeSH-major] Brain Neoplasms / surgery. Cranial Irradiation. Neoplasms, Second Primary. Neuroectodermal Tumors, Primitive / surgery. Precursor Cell Lymphoblastic Leukemia-Lymphoma / radiotherapy
  • [MeSH-minor] Adolescent. Chemotherapy, Adjuvant. Combined Modality Therapy. Humans. Male. Neurosurgical Procedures. Radiotherapy Dosage

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  • (PMID = 16001813.001).
  • [ISSN] 0301-2603
  • [Journal-full-title] No shinkei geka. Neurological surgery
  • [ISO-abbreviation] No Shinkei Geka
  • [Language] jpn
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Japan
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10. Saran F, Baumert BG, Creak AL, Warrington AP, Ashley S, Traish D, Brada M: Hypofractionated stereotactic radiotherapy in the management of recurrent or residual medulloblastoma/PNET. Pediatr Blood Cancer; 2008 Mar;50(3):554-60
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  • [Title] Hypofractionated stereotactic radiotherapy in the management of recurrent or residual medulloblastoma/PNET.
  • PURPOSE: To evaluate the efficacy and toxicity of hypofractionated stereotactic radiotherapy in the management of locally recurrent or residual central nervous system (CNS) primitive neuroectodermal tumors (PNETs).
  • PATIENTS AND METHODS: Between 1991 and 2005, 12 patients with locally recurrent medulloblastoma and two patients with residual supratentorial PNET were treated with hypofractionated stereotactic conformal radiotherapy (SCRT).
  • Nine of 12 patients underwent resection at recurrence and 13 patients received at least one cycle of chemotherapy prior to SCRT.
  • All received focal SCRT (30-40 Gy/6-8 #) using non-coplanar arcs (n = 6) or fixed conformal non-coplanar fields (n = 8).
  • Causes of death were recurrent CNS disease (n = 7), herpes encephalitis (n = 1), and metastatic PNET outside the CNS (n = 1).
  • CONCLUSION: Hypofractionated SCRT provides effective local control with acceptable toxicity for patients with recurrent localized PNET.
  • However, overall long-term disease control is rare and limited by the occurrence of CSF mediated relapses, which thus could benefit from intensive systemic chemotherapy as part of the primary relapse strategy even in local recurrences.
  • Larger multi-national studies will be necessary to assess the value of such combined treatment approaches.
  • [MeSH-major] Cerebellar Neoplasms / radiotherapy. Dose Fractionation. Medulloblastoma / radiotherapy. Neuroectodermal Tumors, Primitive / radiotherapy. Radiotherapy, Conformal / methods. Supratentorial Neoplasms / radiotherapy
  • [MeSH-minor] Adolescent. Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Male. Neoplasm Recurrence, Local / radiotherapy. Neoplasm, Residual. Palliative Care. Retrospective Studies. Stereotaxic Techniques

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  • [Copyright] (c) 2007 Wiley-Liss, Inc.
  • (PMID = 17941071.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Evaluation Studies; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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11. Rosenfeld A, Kletzel M, Duerst R, Jacobsohn D, Haut P, Weinstein J, Rademaker A, Schaefer C, Evans L, Fouts M, Goldman S: A phase II prospective study of sequential myeloablative chemotherapy with hematopoietic stem cell rescue for the treatment of selected high risk and recurrent central nervous system tumors. J Neurooncol; 2010 Apr;97(2):247-55
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  • [Title] A phase II prospective study of sequential myeloablative chemotherapy with hematopoietic stem cell rescue for the treatment of selected high risk and recurrent central nervous system tumors.
  • High risk/recurrent CNS tumors have a poor prognosis.
  • We studied tandem high dose chemotherapy (HDC) with hematopoietic progenitor stem cell rescues (HPCR) as potentially curative therapy.
  • Diagnoses were medulloblastoma (n = 9), germ cell tumor (n = 4), high grade astrocytoma (n = 2), supratentorial PNET (n = 1), pineoblastoma (n = 2), or papillary meningioma (n = 1).
  • Toxicity was significant with six treatment related deaths including four with veno-occlusive disease.
  • This regimen of sequential HDC/HPCR in high risk/recurrent CNS tumor patients is not feasible due to toxicity.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Central Nervous System Neoplasms / therapy. Hematopoietic Stem Cell Transplantation / methods
  • [MeSH-minor] Adolescent. Carboplatin / administration & dosage. Carboplatin / adverse effects. Child. Child, Preschool. Combined Modality Therapy. Cyclophosphamide / administration & dosage. Etoposide / administration & dosage. Etoposide / adverse effects. Female. Humans. Infant. Kaplan-Meier Estimate. Male. Melphalan / administration & dosage. Melphalan / adverse effects. Thiotepa / administration & dosage. Thiotepa / adverse effects. Transplantation Conditioning

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  • (PMID = 19768658.001).
  • [ISSN] 1573-7373
  • [Journal-full-title] Journal of neuro-oncology
  • [ISO-abbreviation] J. Neurooncol.
  • [Language] eng
  • [Publication-type] Clinical Trial, Phase II; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 6PLQ3CP4P3 / Etoposide; 8N3DW7272P / Cyclophosphamide; 905Z5W3GKH / Thiotepa; BG3F62OND5 / Carboplatin; Q41OR9510P / Melphalan
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12. Mikami Y, Nakajima M, Hashimoto H, Irei I, Matsushima T, Kawabata S, Manabe T: Primary pulmonary primitive neuroectodermal tumor (PNET). A case report. Pathol Res Pract; 2001;197(2):113-119; discussion 121-2
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  • [Title] Primary pulmonary primitive neuroectodermal tumor (PNET). A case report.
  • We describe a rare case of a primary primitive neuroectodermal tumor (PNET) in the lung of a 17-year-old girl.
  • Grossly, the tumor, located in the right lower lobe, was relatively well-circumscribed and whitish to yellowish in color with scattered hemorrhagic necrosis.
  • Microscopically, the tumor was composed of ovoid to polygonal cells with a high nuclear to cytoplasmic ratio and relatively scant cytoplasm, arranged in solid sheets with intervening fine fibrovascular stroma.
  • Immunohistochemically, the tumor was positive for the MIC2 gene product, whereas AE1/AE3, CAM5.2, and a variety of neuroendocrine markers such as chromogranin A, synaptophysin, and ProGRP, were negative.
  • Three months after the lobectomy, recurrent tumors were noted in the mediastinum and right thoracic wall, and she died despite combined chemotherapy and radiation therapy.
  • However, the reverse transcriptase-polymerase chain reaction (RT-PCR) demonstrated EWS/FLI-1 fusion transcripts, confirming the histopathologic diagnosis of PNET.
  • This case indicates that the primary pulmonary PNET is a highly aggressive neoplasm occurring at a young age, and should prompt combined systemic chemotherapy, even though it is organ-confined.
  • [MeSH-major] Lung Neoplasms / pathology. Neuroectodermal Tumors, Primitive, Peripheral / pathology
  • [MeSH-minor] Adolescent. Carcinoma, Small Cell / diagnosis. Combined Modality Therapy. DNA Primers / chemistry. DNA, Neoplasm / analysis. Diagnosis, Differential. Fatal Outcome. Female. Humans. Karyotyping. Oncogene Proteins, Fusion / genetics. Proto-Oncogene Protein c-fli-1. RNA-Binding Protein EWS. Radiography, Thoracic. Reverse Transcriptase Polymerase Chain Reaction. Tomography, X-Ray Computed. Transcription Factors / genetics. Tumor Cells, Cultured

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  • (PMID = 11261815.001).
  • [ISSN] 0344-0338
  • [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 / DNA Primers; 0 / DNA, Neoplasm; 0 / EWS-FLI fusion protein; 0 / Oncogene Proteins, Fusion; 0 / Proto-Oncogene Protein c-fli-1; 0 / RNA-Binding Protein EWS; 0 / Transcription Factors
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13. Boiardi A, Silvani A, Eoli M, Fariselli L, Zappacosta B, Salmaggi A: Embryonal tumors in the adult population: implications in therapeutic planning. Neurol Sci; 2000 Feb;21(1):23-30
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  • [Title] Embryonal tumors in the adult population: implications in therapeutic planning.
  • The natural history of neuroectodermal tumors is still debated as far as prognostic factors are concerned; the same uncertainty applies to the optimal radiotherapy schedule and even more to the presumptive additive effect of chemotherapy.
  • The rarity of these tumors and the heterogeneity of management make interpretation of literature data also more difficult.
  • We evaluated clinical course in a cohort of 39 patients, including 31 with medulloblastoma (MB) and 8 with primitive neuroectodermal tumors (PNET).
  • All patients were treated with radiotherapy, a standardized chemotherapy protocol including PCV scheme, and a second-line chemotherapy with cisplatin and etoposide (VP16) at recurrence.
  • In 27 patients, intrathecal chemotherapy was also delivered.
  • Overall, PNET had a worse outcome as compared to MB: median survival times were 42.8 vs. 92.6 months, respectively (p = 0.05).
  • No significant difference in disease-free period was found between patients of different age, desmoplastic variant, tumor localization, or extent of surgery.
  • Systemic or intrathecal chemotherapy did not influence progression-free survival (PFS).
  • However, in the majority of chemotherapy-treated patients, a low-dose craniospinal radiotherapy was also delivered.
  • This combination of treatments may have avoided the expected increased percentage of failure.
  • Moreover, more than half of recurrent patients had a partial response to chemotherapy that extended survival for approximately 3 years.
  • Repeated surgery and chemotherapy at recurrence favorably influenced survival time.
  • [MeSH-major] Brain Neoplasms / drug therapy. Cerebellar Neoplasms / drug therapy. Medulloblastoma / drug therapy. Neuroectodermal Tumors, Primitive / drug therapy. Patient Care Planning
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Cohort Studies. Combined Modality Therapy. Female. Humans. Male. Middle Aged. Neoplasm Recurrence, Local / drug therapy. Retreatment. Survival Analysis

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  • (PMID = 10938199.001).
  • [ISSN] 1590-1874
  • [Journal-full-title] Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
  • [ISO-abbreviation] Neurol. Sci.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] ITALY
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14. Terasaki M, Bouffet E, Katsuki H, Fukushima S, Shigemori M: Pilot trial of the rate of response, safety, and tolerability of temozolomide and oral VP-16 in patients with recurrent or treatment-induced malignant central nervous system tumors. Surg Neurol; 2008 Jan;69(1):46-50
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  • [Title] Pilot trial of the rate of response, safety, and tolerability of temozolomide and oral VP-16 in patients with recurrent or treatment-induced malignant central nervous system tumors.
  • BACKGROUND: The aim of this study was to determine the response and toxicity of patients with recurrent or treatment-induced brain tumors to TMZ and oral VP-16.
  • METHODS: Eleven patients with recurrent or treatment-induced malignant CNS tumors, including treatment-induced PNET (in 1 patient), brainstem glioma (in 3 patients; 1 with treatment-induced, 2 with recurrence), recurrent anaplastic astrocytoma (in 3 patients), and recurrent glioblastoma (in 4 patients) were evaluated in a pilot study of TMZ and oral VP-16 chemotherapy.
  • RESULTS: None experienced major acute toxicity related to TMZ and oral VP-16 during a total of 52 treatment courses.
  • Five (45%) of 11 patients showed a PR to treatment.
  • Among the 11 patients enrolled, 7 patients are alive with disease at a median of 9 months from time of study entry.
  • The histologic subtype of the tumor, its location, and its maximum response to chemotherapy did not have an impact on the duration of disease control.
  • CONCLUSION: This limited pilot study confirms the innocuousness and the activity of the combination of TMZ and oral VP-16 in recurrent malignant brain tumors.
  • [MeSH-major] Antineoplastic Agents / administration & dosage. Brain Neoplasms / drug therapy. Dacarbazine / analogs & derivatives. Etoposide / administration & dosage. Neoplasm Recurrence, Local / drug therapy. Neoplasms, Neuroepithelial / drug therapy. Neoplasms, Second Primary / drug therapy
  • [MeSH-minor] Administration, Oral. Adolescent. Adult. Aged. Drug Therapy, Combination. Female. Humans. Male. Middle Aged. Pilot Projects. Treatment Outcome

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  • (PMID = 18054615.001).
  • [ISSN] 0090-3019
  • [Journal-full-title] Surgical neurology
  • [ISO-abbreviation] Surg Neurol
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 6PLQ3CP4P3 / Etoposide; 7GR28W0FJI / Dacarbazine; 85622-93-1 / temozolomide
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15. Chow WA, Chu P, Chung V, Lawrence J, Garcia D, Doroshow JH: Imatinib mesylate therapy for recurrent Ewing's family of tumors (EFT). J Clin Oncol; 2004 Jul 15;22(14_suppl):9054

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  • [Title] Imatinib mesylate therapy for recurrent Ewing's family of tumors (EFT).
  • : 9054 Background: The prognosis for patients with recurrent Ewing's sarcoma (ES) and primitive neuroectodermal tumors (PNET) remains poor.
  • Novel therapies are urgently needed.
  • Based upon these findings, we treated a patient with recurrent EFT that expressed both c-Kit and PDGFR-α with imatinib mesylate.
  • CT scan in October, 2002 revealed a large retroperitoneal mass, extensive retroperitoneal adenopathy, and ascites; a biopsy revealed PNET.
  • Chemotherapy with vincristine, doxorubicin and cyclophosphamide, altenating with ifosfamide and etoposide was initiated.
  • Progression was documented after 11 cycles of chemotherapy.
  • IHC of the original tumor revealed 3+/3 IHC positivity for both c-Kit and PDGFR-α.
  • CONCLUSIONS: This case report demonstrates that imatinib mesylate was an effective alternative therapy for this patient with recurrent EFT whose tumor expressed the c-Kit and PDGFR-α TKRs.

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  • (PMID = 28014099.001).
  • [ISSN] 1527-7755
  • [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] Journal Article
  • [Publication-country] United States
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16. Partap S, Murphy PA, Vogel H, Barnes PD, Edwards MS, Fisher PG: Efficacy and tolerability of intrathecal liposomal cytarabine for central nervous system embryonal tumors. J Clin Oncol; 2009 May 20;27(15_suppl):2064

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  • [Title] Efficacy and tolerability of intrathecal liposomal cytarabine for central nervous system embryonal tumors.
  • : 2064 Background: Liposomal cytarabine (DepoCyt) is a sustained-release intrathecal (IT) preparation of cytarabine, formulated by encapsulating the drug in spherical aqueous chambers within a lipid matrix.
  • While proven effective in lymphomatous meningitis, this drug has shown some activity in medulloblastoma (MB) with spinal metastases in limited pediatric phase I study.
  • METHODS: We reviewed all patients at our institution treated with liposomal cytarabine for primary central nervous system (CNS) embryonal tumors-MB, primitive neuroectodermal tumor (PNET), and atypical teratoid rhabdoid tumor (ATRT).
  • RESULTS: A cohort of 17 patients were treated with liposomal cytarabine at diagnosis of CNS embryonal tumor (2 PNET, 3 ATRT) or relapse (12 MB [7 average-risk, 5 high-risk]); nine had leptomeningeal metastases.
  • Drug was dosed at 2 mg/kg up to 50, every 2 weeks to monthly, along with dexamethasone.
  • Concurrent systemic chemotherapy was given in 16 patients.
  • A total of 102 doses were administered (lumbar IT 76, Ommaya intraventricular 36) with a mean of six treatments (range 1-16).
  • No patient developed malignant CSF cytology while receiving liposomal cytarabine.
  • Ten patients developed progressive disease and died, with only one later recurrence in the spinal fluid.
  • All patients with neoplastic meningitis cleared malignant cells from their spinal fluid after treatment with IT liposomal cytarabine and systemic chemotherapy.
  • Our findings warrant a phase II trial of liposomal cytarabine in newly diagnosed or recurrent CNS embryonal tumors.

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  • (PMID = 27964690.001).
  • [ISSN] 1527-7755
  • [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] Journal Article
  • [Publication-country] United States
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17. Plotkin M, Amthauer H, Eisenacher J, Wurm R, Michel R, Wust P, Stockhammer F, Röttgen R, Gutberlet M, Ruf J, Felix R: Value of 123I-IMT SPECT for diagnosis of recurrent non-astrocytic intracranial tumours. Neuroradiology; 2005 Jan;47(1):18-26
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  • [Title] Value of 123I-IMT SPECT for diagnosis of recurrent non-astrocytic intracranial tumours.
  • The value of single-photon emission tomography (SPECT) using iodine-123-alpha-methyl-tyrosine (IMT) for the diagnosis of recurrent or residual gliomas is well established.
  • The study included 22 patients with suspected recurrent intracranial tumours of non-astrocytic origin (12 brain metastases, one supratentorial primitive neuroendocrine tumour (PNET), one rhabdoid tumour, two clivus chordomas, three ependymomas, two pituitary tumours, one anaplastic meningioma) who had previously been treated by surgery and/or radio/chemotherapy.
  • We concluded that the IMT-SPECT is a promising complementary imaging tool for the detection of recurrences of non-astrocytic intracranial tumours and their distinguishing from treatment-induced changes.
  • The limitation of the IMT-SPECT is its low sensitivity for the detection of small lesions.
  • [MeSH-major] Brain Neoplasms / diagnostic imaging. Iodine Radioisotopes. Methyltyrosines. Neoplasm Recurrence, Local / diagnostic imaging. Radiopharmaceuticals. Tomography, Emission-Computed, Single-Photon / methods
  • [MeSH-minor] Adolescent. Adult. Aged. Chordoma / diagnostic imaging. Ependymoma / diagnostic imaging. False Negative Reactions. False Positive Reactions. Female. Follow-Up Studies. Glioma / diagnostic imaging. Humans. Magnetic Resonance Imaging. Male. Meningioma / diagnostic imaging. Middle Aged. Neuroendocrine Tumors / diagnostic imaging. Pituitary Neoplasms / diagnostic imaging. Retrospective Studies. Rhabdoid Tumor / diagnostic imaging. Sensitivity and Specificity. Supratentorial Neoplasms / diagnostic imaging

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  • (PMID = 15630586.001).
  • [ISSN] 0028-3940
  • [Journal-full-title] Neuroradiology
  • [ISO-abbreviation] Neuroradiology
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Iodine Radioisotopes; 0 / Methyltyrosines; 0 / Radiopharmaceuticals; A77N8J5H5T / 3-iodo-alpha-methyltyrosine
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18. Casella R, Moch H, Rochlitz C, Meier V, Seifert B, Mihatsch MJ, Gasser TC: Metastatic primitive neuroectodermal tumor of the kidney in adults. Eur Urol; 2001 May;39(5):613-7
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  • [Title] Metastatic primitive neuroectodermal tumor of the kidney in adults.
  • OBJECTIVE: Primitive neuroectodermal tumors (PNET) of the kidney are rare and highly aggressive malignancies.
  • METHODS: The records of 2 patients (30-year-old female and 32-year-old male) with metastatic PNET of the kidney were reviewed and our data compared with the literature.
  • RESULTS: Neither clinical evaluation nor radiological methods allowed to distinguish PNET from renal cell carcinoma.
  • Immunohistochemistry revealed strong positivity for CD99 in tumor 1 and weak positivity for NSE and vimentin in both tumors.
  • In tumor 2, EWS/FLI1 translocation was detected by RT-PCR.
  • Patient 1 underwent nephrectomy, seven cycles of polychemotherapy, two cycles of high-dose chemotherapy, autologous bone marrow rescue, radiotherapy of suspicious skeletal foci and is without evidence of recurrent disease 28 months after therapy.
  • Patient 2 underwent six cycles of polychemotherapy, nephrectomy, high-dose chemotherapy with cyclophosphamide and abdominal radiotherapy.
  • Because of relapse high-dose chemotherapy with stem cell rescue was not performed.
  • CONCLUSIONS: The diagnosis of renal PNET must be considered in young patients with renal neoplasm, particularly those with advanced disease at presentation.
  • Achieving exact diagnosis has important clinical consequences because polychemotherapy and high-dose chemotherapy may lead to dramatic tumor reduction or even complete remission.
  • [MeSH-major] Kidney Neoplasms / pathology. Neuroectodermal Tumors, Primitive / pathology
  • [MeSH-minor] Adult. Antigens, CD / analysis. Cell Adhesion Molecules / analysis. Combined Modality Therapy. Fatal Outcome. Female. Humans. Liver Neoplasms / secondary. Lung Neoplasms / secondary. Male

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  • (PMID = 11464048.001).
  • [ISSN] 0302-2838
  • [Journal-full-title] European urology
  • [ISO-abbreviation] Eur. Urol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Antigens, CD; 0 / CD99 protein, human; 0 / Cell Adhesion Molecules
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19. Burzynski SR, Weaver RA, Janicki T, Szymkowski B, Jurida G, Khan M, Dolgopolov V: Long-term survival of high-risk pediatric patients with primitive neuroectodermal tumors treated with antineoplastons A10 and AS2-1. Integr Cancer Ther; 2005 Jun;4(2):168-77
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  • [Title] Long-term survival of high-risk pediatric patients with primitive neuroectodermal tumors treated with antineoplastons A10 and AS2-1.
  • Primitive neuroectodermal tumors (PNETs) are usually successfully treated with craniospinal radiation and chemotherapy; however, difficulties with standard treatment can be encountered in very young children, in adult patients at high risk of complication from standard treatment, and in patients with recurrent tumors.
  • Thirteen children, either with recurrent disease or high risk, were treated in phase II studies with antineoplastons (ANP).
  • Medulloblastoma was diagnosed in 8 patients, pineoblastoma in 3 patients, and other PNET in 2 patients.
  • Previous treatments included surgery in 12 patients (1 had biopsy only, suboccipital craniotomy), chemotherapy in 6 patients, and radiation therapy in 6 patients.
  • Six patients had not received prior chemotherapy or radiation.
  • The treatment consisted of intravenous infusions of 2 formulations of ANP, A10 and AS2-1, and was administered for an average of 20 months.
  • Six patients (46%) survived more than 5 years from initiation of ANP; 5 were not treated earlier with radiation therapy or chemotherapy.
  • The percentage of patients' response is lower than for standard treatment of favorable PNET, but long-term survival in poor-risk cases and reduced toxicity makes ANP promising for very young children, patients at high risk of complication of standard therapy, and patients with recurrent tumors.
  • [MeSH-major] Antineoplastic Agents / administration & dosage. Benzeneacetamides / administration & dosage. Brain Neoplasms / drug therapy. Glutamine / analogs & derivatives. Neuroectodermal Tumors, Primitive / drug therapy. Phenylacetates / administration & dosage. Piperidones / administration & dosage
  • [MeSH-minor] Child, Preschool. Disease Progression. Drug Combinations. Drug Therapy, Combination. Female. Humans. Infant. Male. Neoplasm Recurrence, Local / prevention & control. Survival Analysis. Treatment Outcome

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  • (PMID = 15911929.001).
  • [ISSN] 1534-7354
  • [Journal-full-title] Integrative cancer therapies
  • [ISO-abbreviation] Integr Cancer Ther
  • [Language] eng
  • [Publication-type] Clinical Trial; Clinical Trial, Phase II; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Benzeneacetamides; 0 / Drug Combinations; 0 / Phenylacetates; 0 / Piperidones; 0RH81L854J / Glutamine; 104624-98-8 / antineoplaston AS 2-1; 91531-30-5 / antineoplaston A10
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20. Baruchel S, Diezi M, Hargrave D, Stempak D, Gammon J, Moghrabi A, Coppes MJ, Fernandez CV, Bouffet E: Safety and pharmacokinetics of temozolomide using a dose-escalation, metronomic schedule in recurrent paediatric brain tumours. Eur J Cancer; 2006 Sep;42(14):2335-42
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  • [Title] Safety and pharmacokinetics of temozolomide using a dose-escalation, metronomic schedule in recurrent paediatric brain tumours.
  • The aims of this study were to determine the maximum tolerated dose (MTD), toxicity and pharmacokinetics of oral temozolomide administered over 42 d in children with recurrent/refractory brain tumours.
  • Three patients in the NHPT group developed grade 3/4 haematological toxicity, 2 experienced dose-limiting toxicity (thrombocytopenia) at 100 mg/m2, and 9/20 developed grade 3 lymphopenia.
  • Responses were observed in 4 patients: 2 complete responses (CR) in medulloblastoma and supratentorial primitive neuroectodermal tumours (PNET), and 2 partial responses (PR) in high-grade glioma, respectively.
  • Overall cumulative exposure was at least 1.5 times higher than in the 5-d administration schedule.
  • [MeSH-major] Antineoplastic Agents, Alkylating / administration & dosage. Brain Neoplasms / drug therapy. Dacarbazine / analogs & derivatives. Neoplasm Recurrence, Local / drug therapy
  • [MeSH-minor] Administration, Oral. Child. Dose-Response Relationship, Drug. Drug Administration Schedule. Feasibility Studies. Female. Humans. Male

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  • (PMID = 16899365.001).
  • [ISSN] 0959-8049
  • [Journal-full-title] European journal of cancer (Oxford, England : 1990)
  • [ISO-abbreviation] Eur. J. Cancer
  • [Language] eng
  • [Publication-type] Clinical Trial, Phase I; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Alkylating; 7GR28W0FJI / Dacarbazine; 85622-93-1 / temozolomide
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21. Brisse H, Servois V, Bouche B, Avni F, Petit P, Thibault F, Zucker JM, Devalck C, Neuenschwander S: Hepatic regenerating nodules: a mimic of recurrent cancer in children. Pediatr Radiol; 2000 Jun;30(6):386-93
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  • [Title] Hepatic regenerating nodules: a mimic of recurrent cancer in children.
  • MATERIALS AND METHODS: Nine children, 2-12 years' old at the time of diagnosis, are described in this retrospective multicentre report.
  • The primary tumours were: nephroblastoma (n = 2), neuroblastoma (n = 2), Ewing's tumour/PNET (n = 2), non-Hodgkin's lymphoma (n = 1), and osteosarcoma (n = 2), treated by surgery (8/9), chemotherapy (9/9), intensive chemotherapy and bone-marrow transplantation (5/9), and radiotherapy (7/9).
  • Three children suffered veno-occlusive disease (VOD) during treatment.
  • RESULTS: Lesions were discovered 15 months to 16 years after completing treatment.
  • CONCLUSION: Pseudometastatic hypervascular hepatic nodules can appear after treatment of a malignant tumour in children.
  • The hypothesis of benign regenerative lesions secondary to treatment and/or VOD is considered.

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  • (PMID = 10876822.001).
  • [ISSN] 0301-0449
  • [Journal-full-title] Pediatric radiology
  • [ISO-abbreviation] Pediatr Radiol
  • [Language] eng
  • [Publication-type] Journal Article; Multicenter Study
  • [Publication-country] GERMANY
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22. Kumar AP, Garcia GE, Orsborn J, Levin VA, Slaga TJ: 2-Methoxyestradiol interferes with NF kappa B transcriptional activity in primitive neuroectodermal brain tumors: implications for management. Carcinogenesis; 2003 Feb;24(2):209-16
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  • [Title] 2-Methoxyestradiol interferes with NF kappa B transcriptional activity in primitive neuroectodermal brain tumors: implications for management.
  • Medulloblastoma (MB) is a primitive neuroectodermal tumor (PNET) of the central nervous system (CNS) and the most common malignant primary brain tumor in children.
  • Currently, poor risk and recurrent MB patients are treated with cytotoxic chemotherapy alone or in combination with surgery and irradiation.
  • In order to improve on therapeutic outcome and reduce toxicity of current treatment strategies, new and novel therapeutic agents are needed for MB patients.
  • 2-ME treatment results in phosphorylation of cdc25C without any significant alterations in the expression of cyclin B1 or p34cdc2.
  • In addition, we observed a decrease in the levels of 14-3-3 proteins following treatment with 2-ME.
  • These results suggest that 2-ME may prove to be a useful therapeutic agent in the treatment of PNET brain tumors such as medulloblastoma.
  • In addition, as 2-ME inhibits growth predominantly through G(2)/M block, it may enhance the effectiveness of radiation therapy.
  • [MeSH-major] Brain Neoplasms / pathology. Estradiol / pharmacology. Medulloblastoma / pathology. NF-kappa B / antagonists & inhibitors. Transcription, Genetic / drug effects
  • [MeSH-minor] Apoptosis / drug effects. Cell Division / drug effects. Humans. Tumor Cells, Cultured

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  • (PMID = 12584169.001).
  • [ISSN] 0143-3334
  • [Journal-full-title] Carcinogenesis
  • [ISO-abbreviation] Carcinogenesis
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / R25 CA49981
  • [Publication-type] 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 / NF-kappa B; 4TI98Z838E / Estradiol; 6I2QW73SR5 / 2-methoxyestradiol
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23. Somers GR, Shago M, Zielenska M, Chan HS, Ngan BY: Primary subcutaneous primitive neuroectodermal tumor with aggressive behavior and an unusual karyotype: case report. Pediatr Dev Pathol; 2004 Sep-Oct;7(5):538-45
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  • [Title] Primary subcutaneous primitive neuroectodermal tumor with aggressive behavior and an unusual karyotype: case report.
  • Primitive neuroectodermal tumor/Ewing sarcoma (PNET/ES) rarely occurs in the skin and subcutaneous tissues.
  • We present a case of a 16-year-old girl with primary cutaneous and subcutaneous PNET/ES of the abdominal wall.
  • Despite wide local excision and chemotherapy, she rapidly developed cranial bone and brain metastases, followed by lung and skeletal metastases, and died shortly thereafter.
  • The recurrent tumor exhibited light microscopic features of a small, round, blue cell tumor with intracytoplasmic glycogen.
  • Cytogenetic analysis of the relapsed tumor showed a complex karyotype: 47,XX,i(1)(q10), der(4)t(4;19) (q33 approximately q35;q13.1), + 8,t(15;17)(q24;p11.2 approximately p12),der(19)t (19;20)(q13.1;p11.2),der(22)t(20;22)(q13;q13).
  • The clinical behavior and atypical and complex cytogenetic abnormalities exhibited by the tumor in this patient are unusual and represent the most aggressive end of the clinical spectrum of cutaneous and subcutaneous PNET/ES.
  • [MeSH-major] Neuroectodermal Tumors, Primitive, Peripheral / secondary. Sarcoma, Ewing / pathology. Soft Tissue Neoplasms / pathology. Subcutaneous Tissue / pathology
  • [MeSH-minor] Adolescent. Biomarkers, Tumor / analysis. Bone Neoplasms / secondary. Brain Neoplasms / metabolism. Brain Neoplasms / secondary. Chromosome Aberrations. Fatal Outcome. Female. Humans. Immunohistochemistry. Lung Neoplasms / secondary. Microscopy, Electron, Transmission. Reverse Transcriptase Polymerase Chain Reaction

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  • (PMID = 15547779.001).
  • [ISSN] 1093-5266
  • [Journal-full-title] Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society
  • [ISO-abbreviation] Pediatr. Dev. Pathol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Biomarkers, Tumor
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24. Cheng NH, Huang HF, Lian LJ, Wu Y: [Ovarian growing teratoma syndrome clinical study of 22 cases]. Zhonghua Fu Chan Ke Za Zhi; 2009 Jun;44(6):426-30
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  • OBJECTIVE: To describe the essential points for the correct diagnosis and best treatment for ovarian growing teratoma syndrome (GTS) developed after surgery and chemotherapy for ovarian immature teratoma.
  • METHODS: Retrospective review of the clinical characteristics and long term follow up results of 22 cases of ovarian GTS to illustrate the unique biological behavior of the tumor and good prognosis of the disease.
  • RESULTS: Pathological examination of the tumors revealed completely benign mature teratoma with G0 grading in 20 cases.
  • The other 2 cases were found to be G0 mature teratoma with concurrent association of malignant somatic cell tumor: carcinoid and primitive neuroectodermal tumor (PNET) respectively.
  • Among the 22 cases of ovarian GTS there are 6 cases with recurrent tumors developed repeatedly, so totally surgical treatments had been performed for 31 times.
  • Time interval in between the development of the ovarian GTS and the initial surgery for their primary immature teratoma is equal to or exceeding one year in 94% (29/31) of the cases.
  • Such a time factor is of high significance for the diagnosis of ovarian GTS.
  • As the benign behavior of the ovarian GTS together with its poor response to chemotherapy have just been recognized in recent years, they were treated as malignant tumors as their original primary immature teratoma before the year of 1987.
  • Postoperative chemotherapy of various kinds was applied.
  • By the year of 1988 postoperative chemotherapy began to be abandoned and since then most of the patients (9/10) had not received postoperative chemotherapy.
  • The rest 2 patients died of the concurrent association of malignant somatic cell tumors with carcinoid and PNET in 0.1 and 0.3 years respectively.
  • CONCLUSIONS: Ovarian GTS is a tumor developed after surgical and chemotherapeutic treatment of malignant ovarian immature teratoma.
  • Pathologic grading of the tumors showed retroconversion of the malignancy of the tumor from G3, G2 or G1 to G0 with good prognosis.
  • The tumor usually remained to be quiescent for long periods of time.
  • But there are also some potential of progressive growth, the tumor may grow to huge size and the recurrent tumor may develop repeatedly for several times more than 10 or 20 years later.
  • Surgical removal should be the main treatment either for the primary or the recurrent tumors.
  • Chemotherapy and radiotherapy are not effective and can do nothing but harm to patients.
  • Only correct knowledge about the benign biological behavior of the ovarian GTS and reasonable therapeutic regimen can have the disease ends with good prognosis.
  • [MeSH-minor] Adolescent. Adult. Carcinoid Tumor / secondary. Child. Female. Follow-Up Studies. Humans. Neoplasm Recurrence, Local / epidemiology. Neoplasm Recurrence, Local / pathology. Prognosis. Retrospective Studies. Syndrome. Treatment Outcome. Young Adult

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  • (PMID = 19953942.001).
  • [ISSN] 0529-567X
  • [Journal-full-title] Zhonghua fu chan ke za zhi
  • [ISO-abbreviation] Zhonghua Fu Chan Ke Za Zhi
  • [Language] chi
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] China
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25. Saenz NC, Hass DJ, Meyers P, Wollner N, Gollamudi S, Bains M, LaQuaglia MP: Pediatric chest wall Ewing's sarcoma. J Pediatr Surg; 2000 Apr;35(4):550-5
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  • [Title] Pediatric chest wall Ewing's sarcoma.
  • BACKGROUND: Chest wall tumors of primitive neuroectodermal origin (PNET, Ewing's sarcoma [ES]) are rare and have a poor prognosis.
  • Multimodality therapy has improved survival results, and long-term survival is possible.
  • Whether adjuvant radiation therapy is uniformly beneficial remains unclear.
  • RESULTS: Twenty consecutive patients with PNET-ES of the chest wall were identified.
  • Initial therapy consisted of biopsy and neoadjuvant chemotherapy followed by chest wall resection in 12 patients.
  • Of the remaining 8 patients, 6 underwent biopsy, followed by chest wall resection and adjuvant chemotherapy, 1 underwent biopsy, chemotherapy, and resection of a lung nodule, and 1 underwent biopsy, chemotherapy, and a laminectomy and decompression procedure.
  • All 20 patients were included in institutional-based trials using multiagent chemotherapy.
  • Fifteen patients received radiation therapy with a median dose of 3,000 cGy.
  • Seven of 11 (64%) survivors had neoadjuvant therapy followed by chest wall resection.
  • Seven of 11 (64%) survivors had radiation therapy.
  • Twelve patients had treatment-related complications, 3 of which were related to surgical resection.
  • There were no survivors among patients with recurrent disease.
  • Only 4 of 9 (44%) patients who died were treated initially with chemotherapy followed by chest wall resection.
  • All but 1 of those that died (89%) received initial radiation therapy.
  • CONCLUSIONS: Long-term survival is possible with ES-PNET after complete chest wall resection.
  • This may be facilitated by neoadjuvant chemotherapy.
  • Long-term survival without radiation therapy is possible, and consideration of radiation therapy should be made on a case-by-case basis.
  • [MeSH-major] Sarcoma, Ewing / mortality. Sarcoma, Ewing / therapy. Thoracic Neoplasms / mortality. Thoracic Neoplasms / therapy
  • [MeSH-minor] Adolescent. Adult. Chemotherapy, Adjuvant. Child. Child, Preschool. Female. Humans. Male. Neoplasm Recurrence, Local. Prognosis. Radiotherapy Dosage. Radiotherapy, Adjuvant. Retrospective Studies. Survival Analysis

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  • (PMID = 10770379.001).
  • [ISSN] 0022-3468
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] UNITED STATES
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26. Flannery T, Kano H, Martin JJ, Niranjan A, Flickinger JC, Lunsford LD, Kondziolka D: Boost radiosurgery as a strategy after failure of initial management of pediatric primitive neuroectodermal tumors. J Neurosurg Pediatr; 2009 Mar;3(3):205-10
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  • [Title] Boost radiosurgery as a strategy after failure of initial management of pediatric primitive neuroectodermal tumors.
  • OBJECT: The aim in this study was to determine the outcomes of boost stereotactic radiosurgery, specifically Gamma Knife surgery (GKS), for recurrent primitive neuroectodermal tumors (PNETs) in children in whom initial multimodality management had failed.
  • METHODS: The authors performed a retrospective analysis of 7 patients who underwent a total of 15 GKS procedures for locally recurrent or metastatic PNETs.
  • All patients received multimodality therapy, including resection, fractionated radiotherapy, and chemotherapy.
  • The mean tumor volume was 3.9 cm(3) (range 1.1-13.1 cm(3)), and an average marginal dose of 14.5 Gy (range 9-20 Gy) was administered.
  • CONCLUSIONS: The GKS modality was a well-tolerated and useful boost strategy in pediatric patients with PNET and a poor prognosis in whom initial multimodality management had failed.
  • [MeSH-major] Brain Neoplasms / surgery. Neuroectodermal Tumors, Primitive / surgery. Radiosurgery
  • [MeSH-minor] Child. Child, Preschool. Combined Modality Therapy. Disease Progression. Female. Humans. Infant. Male. Reoperation. Retrospective Studies. Salvage Therapy. Treatment Outcome. Tumor Burden

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  • [CommentIn] J Neurosurg Pediatr. 2009 Mar;3(3):203; discussion 204 [19267519.001]
  • (PMID = 19338466.001).
  • [ISSN] 1933-0707
  • [Journal-full-title] Journal of neurosurgery. Pediatrics
  • [ISO-abbreviation] J Neurosurg Pediatr
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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27. Fouladi M, Nicholson HS, Zhou T, Laningham F, Helton KJ, Holmes E, Cohen K, Speights RA, Wright J, Pollack IF, Children's Oncology Group: A phase II study of the farnesyl transferase inhibitor, tipifarnib, in children with recurrent or progressive high-grade glioma, medulloblastoma/primitive neuroectodermal tumor, or brainstem glioma: a Children's Oncology Group study. Cancer; 2007 Dec 1;110(11):2535-41
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  • [Title] A phase II study of the farnesyl transferase inhibitor, tipifarnib, in children with recurrent or progressive high-grade glioma, medulloblastoma/primitive neuroectodermal tumor, or brainstem glioma: a Children's Oncology Group study.
  • BACKGROUND: An open-label Phase II study of tipifarnib was conducted to evaluate its safety and efficacy in children with recurrent or refractory medulloblastoma (MB)/primitive neuroectodermal tumor (PNET), high-grade glioma (HGG), and diffuse intrinsic brainstem glioma (BSG).
  • METHODS: Between January 2004 and July 2005, patients were enrolled and stratified as follows: Stratum 1, recurrent or refractory MB/PNET; Stratum 2, recurrent or refractory HGG; and Stratum 3, recurrent or refractory BSG.
  • No responses were observed in 15 patients with MB/PNET.
  • The 6-month progression-free survival rate (+/-standard deviation) was 14%+/-6% for HGG, 6%+/-6% for MB/PNET and 3%+/-3% for BSG.
  • CONCLUSIONS: Tipifarnib tolerated well but had little activity as a single agent in children with recurrent central nervous system malignancies.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Brain Neoplasms / drug therapy. Brain Stem Neoplasms / drug therapy. Glioma / drug therapy. Medulloblastoma / drug therapy. Quinolones / therapeutic use
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Disease Progression. Female. Humans. Male. Neuroectodermal Tumors / drug therapy. Treatment Outcome


28. Karseladze AI, Filipova NA, Navarro S, Llombart-Bosch A: Primitive neuroectodermal tumor of the uterus. A case report. J Reprod Med; 2001 Sep;46(9):845-8
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  • [Title] Primitive neuroectodermal tumor of the uterus. A case report.
  • BACKGROUND: Primitive neuroectodermal tumors (PNETs) of the uterus are very rare.
  • The curettage specimen was interpreted as poorly differentiated sarcoma.
  • Histologically the tumor was composed of uniform, rounded, oval and sometimes spindle shaped cells with a narrow rim of eosinophilic cytoplasm.
  • The cells were positive for neurogenic marker protein gene product, neuron-specific enolase and Ewing's sarcoma-related HBA-71.
  • A diagnosis of PNET was rendered.
  • The patient received combined therapy, external radiation to the pelvis and chemotherapy.
  • Four years later she was alive, without signs of recurrent tumor.
  • CONCLUSION: In spite of the generally recognized aggressive behavior of PNET, it can have a long disease-free survival rate when appropriately treated.
  • [MeSH-major] Neuroectodermal Tumors, Primitive, Peripheral / pathology. Uterine Neoplasms / pathology
  • [MeSH-minor] Adolescent. Antibodies, Neoplasm / isolation & purification. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Combined Modality Therapy. Diagnosis, Differential. Female. Humans. Hysterectomy. Immunohistochemistry

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  • (PMID = 11584489.001).
  • [ISSN] 0024-7758
  • [Journal-full-title] The Journal of reproductive medicine
  • [ISO-abbreviation] J Reprod Med
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antibodies, Neoplasm
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29. Cohen DD, Zorn K, Bernard C, Rajan R, Kuzmarov IW: A para-testicular primitive neuroectodermal tumor in an adult: a case report and literature review. Can J Urol; 2000 Aug;7(4):1081-4
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  • [Title] A para-testicular primitive neuroectodermal tumor in an adult: a case report and literature review.
  • OBJECTIVE: The authors describe the salient clinical, radiologic and histopathologic features of an extremely rare para-testicular primitive neuroectodermal tumor in a 25 year-old man.
  • INTERVENTION: Excisional biopsy of the tumor en bloc was performed.
  • Adjuvant VAdriaC-based chemotherapy (Vincristine, Doxorubicin, and Cyclophosphamide) was given post-operatively.
  • MAIN OUTCOME MEASURES: Histopathologic examination and immunohistochemical studies were performed on formaldehyde-fixed, paraffin-embedded tumor tissue.
  • RESULTS: Histologic examination showed an undifferentiated small cell tumor.
  • The tumor cells stained positively with MIC-2, a marker specific for primitive neuroectodermal tumors.
  • The patient is 12 months post surgery and has completed adjuvant chemotherapy with no evidence of recurrent disease.
  • CONCLUSIONS: This highly unusual, peripheral primitive neuroectodermal tumor should be considered in the differential diagnosis of undifferentiated small cell neoplasms of the genitourinary system in adults, from the kidney to the testicle.
  • We present a patient with a PNET treated based on a Ewing's family of tumors protocol.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Neuroectodermal Tumors, Primitive, Peripheral / pathology. Testicular Neoplasms / pathology

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  • (PMID = 11109079.001).
  • [ISSN] 1195-9479
  • [Journal-full-title] The Canadian journal of urology
  • [ISO-abbreviation] Can J Urol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] Canada
  • [Chemical-registry-number] 0 / Antineoplastic Agents
  • [Number-of-references] 28
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30. Hawkins DS, Bradfield S, Whitlock JA, Krailo M, Franklin J, Blaney SM, Adamson PC, Reaman G: Topotecan by 21-day continuous infusion in children with relapsed or refractory solid tumors: a Children's Oncology Group study. Pediatr Blood Cancer; 2006 Nov;47(6):790-4
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  • [Title] Topotecan by 21-day continuous infusion in children with relapsed or refractory solid tumors: a Children's Oncology Group study.
  • PURPOSE: The Children's Oncology Group conducted a phase II trial of 21-day continuous infusion topotecan to determine the response rate in pediatric patients with recurrent or refractory malignant solid tumors.
  • PROCEDURE: Patients with Ewing sarcoma family of tumors (ESFT), osteosarcoma (OS), soft tissue sarcomas (STS), medulloblastoma (MB)/primitive neuroectodermal tumor (PNET), astrocytoma, or neuroblastoma (NB) recurrent or refractory to conventional therapy, measurable disease, and adequate organ function were treated with topotecan 0.3 mg/m2/day by continuous intravenous infusion for 21 consecutive days, followed by 7 days without therapy prior to response assessment.
  • RESULTS: Fifty-five patients were enrolled; two were ineligible, two were removed from protocol therapy prior to evaluation for response, and one was inevaluable for response, leaving 53 and 50 patients evaluable for toxicity and response, respectively.
  • There were insufficient patients enrolled to determine the response rate for the MB/PNET, astrocytoma, and NB strata.
  • The most common Grade 3 or 4 toxicities during the first course of therapy were thrombocytopenia (12/53), neutropenia (8/53), and fatigue (7/53).
  • CONCLUSION: Intravenous topotecan by 21-day continuous infusion is tolerable in pediatric patients with recurrent or refractory solid tumors.
  • [MeSH-major] Astrocytoma / drug therapy. Neoplasm Recurrence, Local / drug therapy. Neuroblastoma / drug therapy. Neuroectodermal Tumors, Primitive / drug therapy. Sarcoma / drug therapy. Topotecan / administration & dosage
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Dose-Response Relationship, Drug. Fatigue / chemically induced. Female. Humans. Infusions, Intravenous. Male. Maximum Tolerated Dose. Neutropenia / chemically induced. Recurrence. Survival Rate. Thrombocytopenia / chemically induced. Time Factors. Treatment Outcome

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  • [Copyright] (c) 2006 Wiley-Liss, Inc.
  • (PMID = 16435380.001).
  • [ISSN] 1545-5009
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA 98543
  • [Publication-type] Clinical Trial, Phase II; Journal Article; Research Support, N.I.H., Extramural
  • [Publication-country] United States
  • [Chemical-registry-number] 7M7YKX2N15 / Topotecan
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31. Oh J, Bilbao JM, Tsao MN, Fazl M, Guiot MC, Del Maestro RF, Perry JR: Recurrent PNET with MGMT methylation responds to temozolomide. Can J Neurol Sci; 2009 Sep;36(5):654-7
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  • [Title] Recurrent PNET with MGMT methylation responds to temozolomide.
  • [MeSH-major] Antineoplastic Agents, Alkylating / therapeutic use. Brain Neoplasms / drug therapy. Brain Neoplasms / genetics. Dacarbazine / analogs & derivatives. Neuroectodermal Tumors, Primitive / drug therapy. Neuroectodermal Tumors, Primitive / genetics

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  • (PMID = 19831139.001).
  • [ISSN] 0317-1671
  • [Journal-full-title] The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques
  • [ISO-abbreviation] Can J Neurol Sci
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Canada
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Alkylating; 7GR28W0FJI / Dacarbazine; 85622-93-1 / temozolomide; EC 2.1.1.63 / O(6)-Methylguanine-DNA Methyltransferase
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