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1. Li Y, Lal B, Kwon S, Fan X, Saldanha U, Reznik TE, Kuchner EB, Eberhart C, Laterra J, Abounader R: The scatter factor/hepatocyte growth factor: c-met pathway in human embryonal central nervous system tumor malignancy. Cancer Res; 2005 Oct 15;65(20):9355-62
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  • [Title] The scatter factor/hepatocyte growth factor: c-met pathway in human embryonal central nervous system tumor malignancy.
  • Embryonal central nervous system (CNS) tumors, which comprise medulloblastoma, are the most common malignant brain tumors in children.
  • In the present study, we show that human embryonal CNS tumor cell lines and surgical tumor specimens express SF/HGF and c-Met.
  • Treatment of medulloblastoma cells with SF/HGF activates c-Met and downstream signal transduction as evidenced by c-Met, mitogen-activated protein kinase, and Akt phosphorylation.
  • SF/HGF induces tumor cell proliferation, anchorage-independent growth, and cell cycle progression beyond the G1-S checkpoint.
  • SF/HGF also protects medulloblastoma cells against apoptosis induced by chemotherapy.
  • SF/HGF gene transfer to medulloblastoma cells strongly enhances the in vivo growth of s.c. and intracranial tumor xenografts.
  • This first characterization establishes SF/HGF:c-Met as a new pathway of malignancy with multifunctional effects in human embryonal CNS tumors.
  • [MeSH-minor] Animals. Cell Adhesion / drug effects. Cell Adhesion / physiology. Cell Cycle / drug effects. Cell Cycle / physiology. Cell Growth Processes / drug effects. Cell Growth Processes / physiology. Cell Line, Tumor. Cyclin-Dependent Kinase 2 / metabolism. Cyclin-Dependent Kinase Inhibitor p27 / metabolism. Humans. Intracellular Signaling Peptides and Proteins / metabolism. Mice. Mitogen-Activated Protein Kinases / metabolism. Neoplasm Transplantation. Phosphorylation. Prognosis. Proto-Oncogene Proteins c-akt / metabolism. RNA, Messenger / biosynthesis. RNA, Messenger / genetics. Transplantation, Heterologous

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  • (PMID = 16230398.001).
  • [ISSN] 0008-5472
  • [Journal-full-title] Cancer research
  • [ISO-abbreviation] Cancer Res.
  • [Language] eng
  • [Grant] United States / NINDS NIH HHS / NS / NS 43987; United States / NINDS NIH HHS / NS / R01 NS032148; United States / NINDS NIH HHS / NS / R01 NS045209
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / CDKN1B protein, human; 0 / Intracellular Signaling Peptides and Proteins; 0 / RNA, Messenger; 147604-94-2 / Cyclin-Dependent Kinase Inhibitor p27; 67256-21-7 / Hepatocyte Growth Factor; EC 2.7.10.1 / Proto-Oncogene Proteins c-met; EC 2.7.11.1 / Proto-Oncogene Proteins c-akt; EC 2.7.11.22 / Cyclin-Dependent Kinase 2; EC 2.7.11.24 / Mitogen-Activated Protein Kinases
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2. Fouladi M, Gururangan S, Moghrabi A, Phillips P, Gronewold L, Wallace D, Sanford RA, Gajjar A, Kun LE, Heideman R: Carboplatin-based primary chemotherapy for infants and young children with CNS tumors. Cancer; 2009 Jul 15;115(14):3243-53
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  • [Title] Carboplatin-based primary chemotherapy for infants and young children with CNS tumors.
  • BACKGROUND: A carboplatin-based chemotherapy regimen was used as primary postoperative therapy in infants with central nervous system (CNS) tumors to limit renal and ototoxicity and to target systemic exposure.
  • METHODS: Fifty-three patients aged <age 3 years with embryonal CNS tumor medulloblastoma (n = 20), ependymoma (EP, n = 21), choroid plexus carcinoma (CPCA, n = 5), and primitive embryonal neoplasms including atypical teratoid rhabdoid tumors (n = 7) were treated with cyclophosphamide, etoposide, and carboplatin.
  • Radiation therapy was used only for residual disease at the end of chemotherapy or disease progression.
  • RESULTS: The response rate after 2 cycles of chemotherapy was 34% (complete response, 13.8%; partial response, 20.7%).
  • Two patients developed late second malignancies; 1 was associated with germline p53 mutation.
  • Five-year survival data are comparable to those reported in other recent studies, including high-dose chemotherapy studies.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Carboplatin / administration & dosage. Central Nervous System Neoplasms / drug therapy
  • [MeSH-minor] Chemotherapy, Adjuvant. Child, Preschool. Combined Modality Therapy. Cyclophosphamide / administration & dosage. Etoposide / administration & dosage. Humans. Infant. Survival Analysis. Time Factors. Treatment Outcome

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  • (PMID = 19484793.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / P30 CA021765; United States / NCI NIH HHS / CA / P30 CA021765-30; United States / NCI NIH HHS / CA / R25 CA023944; United States / NCI NIH HHS / CA / CA21765
  • [Publication-type] Clinical Trial, Phase II; Journal Article; Multicenter Study; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 6PLQ3CP4P3 / Etoposide; 8N3DW7272P / Cyclophosphamide; BG3F62OND5 / Carboplatin
  • [Other-IDs] NLM/ NIHMS124374; NLM/ PMC4307774
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3. Yoo KH, Lee SH, Lee J, Sung KW, Jung HL, Koo HH, Lim DH, Kim JH, Shin HJ: Improved outcome of central nervous system germ cell tumors: implications for the role of risk-adapted intensive chemotherapy. J Korean Med Sci; 2010 Mar;25(3):458-65
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  • [Title] Improved outcome of central nervous system germ cell tumors: implications for the role of risk-adapted intensive chemotherapy.
  • To determine the impact of treatment protocols on the outcome of central nervous system germ cell tumors (CNS-GCTs), we reviewed the medical records of 53 patients who received front-line chemotherapy from September 1997 to September 2006.
  • Patients from different time periods were divided into 3 groups according to the chemotherapy protocols.
  • Group 1 (n=19) received 4 cycles of chemotherapy comprising cisplatin, etoposide and bleomycin.
  • Group 2 (n=16) and group 3 (n=18) received 4 cycles of chemotherapy with cisplatin, etoposide, cyclophosphamide and vincristine in the former and with carboplatin, etoposide, cyclophosphamide and bleomycin in the latter.
  • Radiotherapy was given after chemotherapy according to the clinical requirements.
  • Our data suggest that risk-adapted intensive chemotherapy may improve the outcome of patients with malignant CNS-GCTs.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Central Nervous System Neoplasms / therapy. Neoplasms, Germ Cell and Embryonal / therapy. Radiotherapy
  • [MeSH-minor] Adolescent. Biomarkers, Tumor / metabolism. Child. Combined Modality Therapy. Disease-Free Survival. Female. Humans. Kaplan-Meier Estimate. Male. Risk Factors. Treatment Outcome. Young Adult

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  • (PMID = 20191048.001).
  • [ISSN] 1598-6357
  • [Journal-full-title] Journal of Korean medical science
  • [ISO-abbreviation] J. Korean Med. Sci.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Korea (South)
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Biomarkers, Tumor
  • [Other-IDs] NLM/ PMC2826748
  • [Keywords] NOTNLM ; Central Nervous System / Drug Therapy / Neoplasms, Germ Cell and Embryonal / Survival
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4. Ferri Niguez B, Martínez-Lage JF, Almagro MJ, Fuster JL, Serrano C, Torroba MA, Sola J: Embryonal tumor with abundant neuropil and true rosettes (ETANTR): a new distinctive variety of pediatric PNET: a case-based update. Childs Nerv Syst; 2010 Aug;26(8):1003-8
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  • [Title] Embryonal tumor with abundant neuropil and true rosettes (ETANTR): a new distinctive variety of pediatric PNET: a case-based update.
  • BACKGROUND: Embryonal central nervous system (CNS) tumors are currently classified into three types: medulloblastoma, atypical rhabdoid/teratoid tumors, and primitive neuroectodermal tumor (PNET).
  • A distinctive subtype of PNET called "embryonal tumor with abundant neuropil and true rosettes" (ETANTR) was reported in 2000.
  • It has been suggested that this neoplasm should be considered as a separate entity.
  • ETANTR is an eminently pediatric tumor that has been reported exclusively in children younger than 4 years.
  • ILLUSTRATIVE CASES: A 9-month-old girl underwent subtotal resection of a brainstem neoplasm.
  • A 23-month-old girl was submitted to surgery for a frontoparietal tumor.
  • Both children were treated with chemotherapy and one with radiotherapy.
  • CONCLUSIONS: By reporting these two new instances of ETANTR, we want to contribute to the knowledge of this highly malignant CNS embryonal neoplasm that occurs only in young children, given its present lethal prognosis, the scarcity of reported cases, and the lack of treatment guidelines.
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Combined Modality Therapy. Fatal Outcome. Female. Humans. Infant. Neurosurgical Procedures. Radiotherapy

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  • (PMID = 20499240.001).
  • [ISSN] 1433-0350
  • [Journal-full-title] Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
  • [ISO-abbreviation] Childs Nerv Syst
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents
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5. Sands SA, Kellie SJ, Davidow AL, Diez B, Villablanca J, Weiner HL, Pietanza MC, Balmaceda C, Finlay JL: Long-term quality of life and neuropsychologic functioning for patients with CNS germ-cell tumors: from the First International CNS Germ-Cell Tumor Study. Neuro Oncol; 2001 07;3(3):174-83
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  • [Title] Long-term quality of life and neuropsychologic functioning for patients with CNS germ-cell tumors: from the First International CNS Germ-Cell Tumor Study.
  • This study evaluated the quality of life and neuropsychologic functioning among patients enrolled between 1989 and 1993 in the First International CNS Germ-Cell Tumor Study.
  • Those who received CNS radiation therapy (n = 29) reported significantly worse physical health, but similar psychosocial health, compared with those treated without radiation.
  • Younger patients diagnosed with CNS germ-cell tumors are at increased risk for psychosocial and physical problems as well as neuropsychologic deficits.
  • Exposure to irradiation adversely affects overall physical functioning, whereas tumor pathology appears to be a salient neurocognitive risk factor.
  • Collaborative and randomized studies are required to further elucidate the late effects arising from factors such as age at diagnosis, tumor histology, level of irradiation therapy, and chemotherapy toxicity among these young and potentially curable patients.
  • [MeSH-major] Central Nervous System Neoplasms / psychology. Intelligence. Neoplasms, Germ Cell and Embryonal / psychology. Quality of Life

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  • (PMID = 11465398.001).
  • [ISSN] 1522-8517
  • [Journal-full-title] Neuro-oncology
  • [ISO-abbreviation] Neuro-oncology
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Other-IDs] NLM/ PMC1920620
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6. da Silva NS, Cappellano AM, Diez B, Cavalheiro S, Gardner S, Wisoff J, Kellie S, Parker R, Garvin J, Finlay J: Primary chemotherapy for intracranial germ cell tumors: results of the third international CNS germ cell tumor study. Pediatr Blood Cancer; 2010 Mar;54(3):377-83
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  • [Title] Primary chemotherapy for intracranial germ cell tumors: results of the third international CNS germ cell tumor study.
  • BACKGROUND: The treatment of central nervous system (CNS) germ cell tumors (GCT) remains controversial.
  • The purpose of this study was to demonstrate efficacy of a chemotherapy only strategy, with less morbidity, when compared to regimens with irradiation.
  • METHODS: Between January 2001 and December 2004 newly diagnosed patients with CNS GCT were treated with one of two risk-tailored chemotherapy regimens.
  • Twenty-five patients aged 4 months to 24.5 years were stratified: Regimen A consisted of 4-6 cycles of carboplatin/etoposide alternating with cyclophosphamide/etoposide for low risk (LR) localized germinoma with normal cerebrospinal fluid (CSF) and serum tumor markers.
  • Seventeen (68%) patients achieved complete radiographic and marker responses after two courses and 19 (76%) after four courses of chemotherapy.
  • CONCLUSION: These intensive chemotherapy regimens proved less effective than irradiation containing regimens.
  • Our results indicate that, at the present time, standard treatment for CNS GCT continues to include irradiation either alone or combined with chemotherapy for pure germinomas and with chemotherapy for those with MMGCT.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / drug therapy. Neoplasms, Germ Cell and Embryonal / drug therapy
  • [MeSH-minor] Adolescent. Adult. Carboplatin / administration & dosage. Carboplatin / adverse effects. Child. Child, Preschool. Combined Modality Therapy. Cyclophosphamide / administration & dosage. Cyclophosphamide / adverse effects. Etoposide / administration & dosage. Etoposide / adverse effects. Female. Humans. Infant. Male. Treatment Outcome. Young Adult

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  • [Copyright] Copyright 2009 Wiley-Liss, Inc.
  • (PMID = 20063410.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 6PLQ3CP4P3 / Etoposide; 8N3DW7272P / Cyclophosphamide; BG3F62OND5 / Carboplatin
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7. Kretschmar C, Kleinberg L, Greenberg M, Burger P, Holmes E, Wharam M: Pre-radiation chemotherapy with response-based radiation therapy in children with central nervous system germ cell tumors: a report from the Children's Oncology Group. Pediatr Blood Cancer; 2007 Mar;48(3):285-91
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  • [Title] Pre-radiation chemotherapy with response-based radiation therapy in children with central nervous system germ cell tumors: a report from the Children's Oncology Group.
  • BACKGROUND: This Phase II study was designed to determine response to chemotherapy and survival after response-based radiation (RT) in children with CNS germ cell tumors.
  • PROCEDURE: Children with germinomas and normal markers received cisplatin 100 mg/m(2) + etoposide, alternating with vincristine + cyclophosphamide (CPM) 2 g/m(2)/d, for four cycles.
  • For germinoma patients in complete response (CR), RT was decreased from 50.4 to 30.6 Gy.
  • High-risk patients received neuraxis RT: 50.4 Gy local + 30.6 Gy neuraxis in CR; 54 Gy local + 36 Gy if less than CR.

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  • [Copyright] (c) 2006 Wiley-Liss, Inc.
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  • (PMID = 16598761.001).
  • [ISSN] 1545-5009
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] ENG
  • [Grant] United States / NCI NIH HHS / CA / P30 CA006973
  • [Publication-type] Clinical Trial, Phase II; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / Chorionic Gonadotropin; 0 / alpha-Fetoproteins; 5J49Q6B70F / Vincristine; 6PLQ3CP4P3 / Etoposide; 8N3DW7272P / Cyclophosphamide; Q20Q21Q62J / Cisplatin
  • [Other-IDs] NLM/ NIHMS582771; NLM/ PMC4086720
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8. Khatua S, Dhall G, O'Neil S, Jubran R, Villablanca JG, Marachelian A, Nastia A, Lavey R, Olch AJ, Gonzalez I, Gilles F, Nelson M, Panigrahy A, McComb G, Krieger M, Fan J, Sposto R, Finlay JL: Treatment of primary CNS germinomatous germ cell tumors with chemotherapy prior to reduced dose whole ventricular and local boost irradiation. Pediatr Blood Cancer; 2010 Jul 15;55(1):42-6
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Treatment of primary CNS germinomatous germ cell tumors with chemotherapy prior to reduced dose whole ventricular and local boost irradiation.
  • BACKGROUND: The purpose of this study was to evaluate a reduced irradiation dose strategy for newly diagnosed primary central nervous system (CNS) germinomas.
  • In 18 patients, chemotherapy was followed by whole ventricular irradiation to 21.6-25.5 Gy with a simultaneous integrated or sequential primary site boost to 30-30.6 Gy.
  • Initial tumor markers for beta-human chorionic gonadotrophin (HCGbeta) and alpha-fetoprotein (AFP) were evaluated in serum and lumbar cerebrospinal fluid (CSF).
  • Neurocognitive function was evaluated periodically following treatment.
  • RESULTS: Eighteen of 20 patients are without evidence of residual or recurrent tumor.
  • Both relapsing patients were subsequently determined to harbor malignant non-germinomatous germ cell tumor (NGGCT).
  • CONCLUSION: Chemotherapy followed by reduced dose whole ventricular and local boost irradiation appears to be effective in patients with localized pure CNS germinoma with evidence of preservation of neurocognitive function.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / therapy. Central Nervous System Neoplasms / therapy. Germinoma / therapy. Neoplasms, Germ Cell and Embryonal / therapy
  • [MeSH-minor] Adolescent. Adult. Carboplatin / adverse effects. Carboplatin / therapeutic use. Child. Child, Preschool. Chorionic Gonadotropin, beta Subunit, Human / blood. Combined Modality Therapy. Dose-Response Relationship, Radiation. Etoposide / adverse effects. Etoposide / therapeutic use. Female. Humans. Infant. Magnetic Resonance Imaging. Male. Retrospective Studies. Young Adult. alpha-Fetoproteins / analysis

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  • (PMID = 20222020.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Chorionic Gonadotropin, beta Subunit, Human; 0 / alpha-Fetoproteins; 6PLQ3CP4P3 / Etoposide; BG3F62OND5 / Carboplatin
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9. Merchant TE, Kiehna EN, Li C, Shukla H, Sengupta S, Xiong X, Gajjar A, Mulhern RK: Modeling radiation dosimetry to predict cognitive outcomes in pediatric patients with CNS embryonal tumors including medulloblastoma. Int J Radiat Oncol Biol Phys; 2006 May 01;65(1):210-21
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  • [Title] Modeling radiation dosimetry to predict cognitive outcomes in pediatric patients with CNS embryonal tumors including medulloblastoma.
  • PURPOSE: Model the effects of radiation dosimetry on IQ among pediatric patients with central nervous system (CNS) tumors.
  • METHODS AND MATERIALS: Pediatric patients with CNS embryonal tumors (n = 39) were prospectively evaluated with serial cognitive testing, before and after treatment with postoperative, risk-adapted craniospinal irradiation (CSI) and conformal primary-site irradiation, followed by chemotherapy.
  • For most models, each Gy of exposure had a similar effect on IQ decline, regardless of dose level.
  • Despite measures to reduce radiation dose and treatment volume, the volume that receives the highest dose continues to have the greatest effect, which supports current volume-reduction efforts.
  • [MeSH-major] Central Nervous System Neoplasms / radiotherapy. Cognition Disorders / etiology. Intelligence / radiation effects. Models, Psychological. Neoplasms, Germ Cell and Embryonal / radiotherapy
  • [MeSH-minor] Child. Child, Preschool. Cranial Irradiation / methods. Dose-Response Relationship, Radiation. Female. Humans. Intelligence Tests. Male. Medulloblastoma / drug therapy. Medulloblastoma / radiotherapy. Neuroectodermal Tumors / drug therapy. Neuroectodermal Tumors / radiotherapy. Prospective Studies. Radiotherapy Dosage. Rhabdoid Tumor / drug therapy. Rhabdoid Tumor / radiotherapy. Supratentorial Neoplasms / drug therapy. Supratentorial Neoplasms / radiotherapy. Temporal Lobe / radiation effects

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  • (PMID = 16472938.001).
  • [ISSN] 0360-3016
  • [Journal-full-title] International journal of radiation oncology, biology, physics
  • [ISO-abbreviation] Int. J. Radiat. Oncol. Biol. Phys.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / R25 CA023944; United States / NCI NIH HHS / CA / CA21765
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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10. Biswas S, Burke A, Cherian S, Williams D, Nicholson J, Horan G, Jefferies S, Williams M, Earl HM, Burnet NG, Hatcher H: Non-pineal supratentorial primitive neuro-ectodermal tumors (sPNET) in teenagers and young adults: Time to reconsider cisplatin based chemotherapy after cranio-spinal irradiation? Pediatr Blood Cancer; 2009 Jul;52(7):796-803
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  • [Title] Non-pineal supratentorial primitive neuro-ectodermal tumors (sPNET) in teenagers and young adults: Time to reconsider cisplatin based chemotherapy after cranio-spinal irradiation?
  • BACKGROUND: Supratentorial PNET (sPNET) are rare CNS tumors of embryonal origin arising in children and adults.
  • The treatment of sPNET for all age groups at our cancer center has been based on the management of medulloblastoma (MB), involving neurosurgical debulking followed by cranio-spinal irradiation (CSI) and systemic chemotherapy.
  • METHODS: Medical records were reviewed to gather demographic and clinical data about all embryonal CNS tumors in children and adults from 2001 to 2007.
  • Tumor pathology, clinical management and survival data were also assessed, particularly as regards those patients who received the Packer chemotherapy regimen for either sPNET or MB.
  • When only considering those patients treated with the Packer chemotherapy regimen, the 5-year OS was 12% for sPNET versus 79% for MB.
  • CONCLUSION: This retrospective study demonstrates that non-pineal sPNET are clinically distinct from MB and are resistant to the Packer chemotherapy regimen.
  • We suggest that it is time to reconsider the use of this regimen in teenage and young adult non-pineal sPNET and to investigate the utility of alternative approaches.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Cerebellar Neoplasms / therapy. Cranial Irradiation. Medulloblastoma / therapy. Neuroectodermal Tumors, Primitive / therapy. Supratentorial Neoplasms / therapy
  • [MeSH-minor] Adolescent. Adult. Child. Cisplatin / administration & dosage. Follow-Up Studies. Humans. Lomustine / administration & dosage. Neoplasm Staging. Retrospective Studies. Survival Rate. Treatment Outcome. Vincristine / administration & dosage. Young Adult

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  • [Copyright] (c) 2009 Wiley-Liss, Inc.
  • (PMID = 19202566.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Clinical Trial, Phase I; Clinical Trial, Phase II; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 5J49Q6B70F / Vincristine; 7BRF0Z81KG / Lomustine; Q20Q21Q62J / Cisplatin
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11. 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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12. Al-Hussain TO, Dababo MA: Posterior fossa tumor in a 2 year-old girl. Brain Pathol; 2009 Apr;19(2):343-6

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  • [Title] Posterior fossa tumor in a 2 year-old girl.
  • We report a case of a 2 year-old girl who presented with three weeks' history of deterioration of walking, then became unable to walk and later she developed projectile vomiting.
  • The tumor was diagnosed as an embryonal tumor with abundant neuropil and true rosettes (ETANTR).
  • The tumor cells in the neuroblastic component were diffusely positive for synaptophysin and CD56, with scattered positive cells for glial fibrillary acidic protein.
  • Our patient developed recurrent disease 6 months after resection and chemotherapy.
  • ETANTR is a very rare aggressive embryonal CNS tumor that combines features of neuroblastoma and ependymoblastoma.
  • [MeSH-major] Infratentorial Neoplasms / pathology. Neoplasms, Germ Cell and Embryonal / pathology

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  • (PMID = 19291003.001).
  • [ISSN] 1750-3639
  • [Journal-full-title] Brain pathology (Zurich, Switzerland)
  • [ISO-abbreviation] Brain Pathol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Switzerland
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13. Yasuda K, Taguchi H, Sawamura Y, Ikeda J, Aoyama H, Fujieda K, Ishii N, Kashiwamura M, Iwasaki Y, Shirato H: Low-dose craniospinal irradiation and ifosfamide, cisplatin and etoposide for non-metastatic embryonal tumors in the central nervous system. Jpn J Clin Oncol; 2008 Jul;38(7):486-92
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  • [Title] Low-dose craniospinal irradiation and ifosfamide, cisplatin and etoposide for non-metastatic embryonal tumors in the central nervous system.
  • OBJECTIVE: The current study was conducted to evaluate the effects of low-dose craniospinal irradiation (CSI) combined with chemotherapy on non-metastatic embryonal tumors in the central nervous system (CNS), including medulloblastoma and supra-tentorial primitive neuroectodermal tumors (ST-PNET).
  • After surgery, the patients < or =5 years old received 18 Gy and the patients >5 years old received 24 Gy CSI.
  • The dose to the primary tumor bed was 39.6-54 Gy.
  • Chemotherapy consisted of ifosfamide, cisplatin and etoposide (ICE chemotherapy).
  • Eight patients received hormone replacement therapy.
  • CONCLUSION: Low-dose CSI and ICE chemotherapy may have a role as a treatment option for a subset of patients with non-metastatic embryonal tumors in the CNS.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Brain Neoplasms / therapy. Cranial Irradiation. Medulloblastoma / therapy. Neuroectodermal Tumors, Primitive / therapy
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Cisplatin / administration & dosage. Combined Modality Therapy. Dose-Response Relationship, Radiation. Etoposide / administration & dosage. Female. Humans. Ifosfamide / administration & dosage. Infant. Male. Radiotherapy Dosage. Supratentorial Neoplasms / pathology. Supratentorial Neoplasms / therapy. Survival Analysis

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  • (PMID = 18573848.001).
  • [ISSN] 1465-3621
  • [Journal-full-title] Japanese journal of clinical oncology
  • [ISO-abbreviation] Jpn. J. Clin. Oncol.
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Japan
  • [Chemical-registry-number] 6PLQ3CP4P3 / Etoposide; Q20Q21Q62J / Cisplatin; UM20QQM95Y / Ifosfamide
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14. Blaney SM, Boyett J, Friedman H, Gajjar A, Geyer R, Horowtiz M, Hunt D, Kieran M, Kun L, Packer R, Phillips P, Pollack IF, Prados M, Heideman R: Phase I clinical trial of mafosfamide in infants and children aged 3 years or younger with newly diagnosed embryonal tumors: a pediatric brain tumor consortium study (PBTC-001). J Clin Oncol; 2005 Jan 20;23(3):525-31
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  • [Title] Phase I clinical trial of mafosfamide in infants and children aged 3 years or younger with newly diagnosed embryonal tumors: a pediatric brain tumor consortium study (PBTC-001).
  • PURPOSE: A phase I trial of intrathecal (IT) mafosfamide was performed to determine the optimal dose, dose-limiting toxicities, and incidence and severity of other toxicities when administered in association with concomitant multiagent systemic chemotherapy to children younger than 3 years with newly diagnosed embryonal tumors.
  • CONCLUSION: The maximum tolerated dose and recommended phase II dose of IT mafosfamide in patients younger than 3 years with newly diagnosed embryonal CNS tumors is 14 mg.
  • A trial to assess the efficacy of regional therapy with IT mafosfamide administered with intensive systemic chemotherapy in children younger than 3 years with primary intracranial embryonal tumors is now in progress.
  • [MeSH-major] Antineoplastic Agents / adverse effects. Antineoplastic Agents / therapeutic use. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / drug therapy. Cyclophosphamide / analogs & derivatives. Cyclophosphamide / therapeutic use. Neoplasms, Germ Cell and Embryonal / drug therapy

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  • (PMID = 15659498.001).
  • [ISSN] 0732-183X
  • [Journal-full-title] Journal of clinical oncology : official journal of the American Society of Clinical Oncology
  • [ISO-abbreviation] J. Clin. Oncol.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA 98007
  • [Publication-type] Clinical Trial; Clinical Trial, Phase I; Journal Article; Multicenter Study; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Analgesics, Opioid; 0 / Antineoplastic Agents; 0 / Glucocorticoids; 5970HH9923 / mafosfamide; 76I7G6D29C / Morphine; 7S5I7G3JQL / Dexamethasone; 8N3DW7272P / Cyclophosphamide
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15. Göbel U, Schneider DT, Teske C, Schönberger S, Calaminus G: Brain metastases in children and adolescents with extracranial germ cell tumor - data of the MAHO/MAKEI-registry. Klin Padiatr; 2010 May;222(3):140-4
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  • [Title] Brain metastases in children and adolescents with extracranial germ cell tumor - data of the MAHO/MAKEI-registry.
  • BACKGROUND: We analyzed 15 children and adolescents with extracranial germ cell tumor (GCT) and brain metastases reported to the MAHO/MAKEI registry.
  • All patients with advanced malignant GCTs received cisplatin-based chemotherapy (overall survival: 0.81+/-0.04 (734/823).
  • RESULTS: 15 patients with brain metastases were reported; in 6 of them at diagnosis and 9 respectively during follow-up (6 weeks-28 months after end of therapy, mean=10 months).
  • In all patients with secondary brain metastases the previously normalised tumor markers AFP and/ or HCG increased again prior to the onset of neurological symptoms.
  • Only 1 of the patients with primary brain metastases survived, whereas 4 of 9 with secondary metastases are in remission after additional treatment.
  • Development of neurological symptoms at initial diagnosis or during follow-up should lead to rapid clinical re-evaluation including CNS imaging and assessment of tumor markers.
  • Treatment of brain metastases includes intensified chemotherapy and surgical resection, irradiation has to be considered in special clinical situations.
  • [MeSH-major] Brain Neoplasms / secondary. Neoplasms, Germ Cell and Embryonal / secondary. Registries
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Biomarkers, Tumor / blood. Child. Child, Preschool. Cisplatin / administration & dosage. Combined Modality Therapy. Cranial Irradiation. Craniotomy. Female. Humans. Infant. Male. Prospective Studies. Risk Factors. Survival Rate

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  • (PMID = 20514616.001).
  • [ISSN] 1439-3824
  • [Journal-full-title] Klinische Pädiatrie
  • [ISO-abbreviation] Klin Padiatr
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; Q20Q21Q62J / Cisplatin
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16. Kim A, Ji L, Balmaceda C, Diez B, Kellie SJ, Dunkel IJ, Gardner SL, Sposto R, Finlay JL: The prognostic value of tumor markers in newly diagnosed patients with primary central nervous system germ cell tumors. Pediatr Blood Cancer; 2008 Dec;51(6):768-73
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  • [Title] The prognostic value of tumor markers in newly diagnosed patients with primary central nervous system germ cell tumors.
  • BACKGROUND: To determine the impact of diagnostic serum and/or cerebrospinal fluid (CSF) alpha-fetoprotein (AFP) and beta-human chorionic gonadotropin (b-HCG) elevations on survival in newly diagnosed patients with central nervous system germ cell tumors (CNS GCT) treated with chemotherapy with the intent to avoid irradiation.
  • PROCEDURE: Seventy-five patients with newly diagnosed CNS GCT enrolled in two sequential internationally conducted clinical trials with serum and CSF AFP and b-HCG levels available from initial diagnosis were retrospectively analyzed.
  • Subjects received platinum based chemotherapy and were followed with serial imaging and tumor marker evaluations.
  • RESULTS: The 5-year overall survival (OS) and event free survival (EFS) for patients with normal tumor markers compared with those with elevated markers at diagnosis was 78% (95% CI 51-91%) versus 60% (95% CI 46-72%) (P = 0.08) and 22% (95% CI 7-43%) versus 28% (95% CI 16-40%) (P = 0.68).
  • The hazard ratio of death for patients with elevated markers was 1.9 times as high as that for those with normal markers (95% CI 0.58-6.5) after adjusting for other baseline characteristics.
  • CONCLUSIONS: Patients with elevated tumor markers appear to have poorer OS independent of tumor histology, although these differences do not reach statistical significance (P < or = 0.05).
  • No differences were observed in EFS between groups likely due to the poor response of chemotherapy only approach to patients with normal markers. b-HCG elevations in biopsy proven germinomas do not seem to alter a patient's prognosis.
  • [MeSH-major] Biomarkers, Tumor / blood. Biomarkers, Tumor / cerebrospinal fluid. Brain Neoplasms / mortality. Neoplasms, Germ Cell and Embryonal / mortality
  • [MeSH-minor] Adolescent. Child. Chorionic Gonadotropin, beta Subunit, Human / blood. Chorionic Gonadotropin, beta Subunit, Human / cerebrospinal fluid. Disease-Free Survival. Female. Humans. Male. Neoplasm Staging. Prognosis. Retrospective Studies. Survival Rate. Treatment Outcome. alpha-Fetoproteins / analysis. alpha-Fetoproteins / cerebrospinal fluid

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  • (PMID = 18802946.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / AFP protein, human; 0 / Biomarkers, Tumor; 0 / Chorionic Gonadotropin, beta Subunit, Human; 0 / alpha-Fetoproteins
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17. Doyle DM, Einhorn LH: Delayed effects of whole brain radiotherapy in germ cell tumor patients with central nervous system metastases. Int J Radiat Oncol Biol Phys; 2008 Apr 1;70(5):1361-4
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  • [Title] Delayed effects of whole brain radiotherapy in germ cell tumor patients with central nervous system metastases.
  • PURPOSE: Central nervous system (CNS) metastases are uncommon in patients with germ cell tumors, with an incidence of 2-3%.
  • CNS metastases have been managed with whole brain radiotherapy (WBRT) and concomitant cisplatin-based combination chemotherapy.
  • Our previous study did not observe serious CNS toxicity (Int J Radiat Oncol Biol Phys 1991;22:17-22).
  • We now report on 5 patients who developed delayed significant CNS toxicity.
  • PATIENTS AND METHODS: We observed 5 patients with delayed CNS toxicity.
  • Of the 5 patients, 3 had CNS metastases at diagnosis and 2 developed relapses with CNS metastases.
  • These 5 patients underwent WBRT to 4,000-5,000 cGy in 18-28 fractions concurrently with cisplatin-based chemotherapy.
  • RESULTS: All 5 patients developed delayed symptoms consistent with progressive multifocal leukoencephalopathy.
  • The median time from WBRT to CNS symptoms was 72 months (range, 9-228).
  • Treatment with surgery and/or steroids had modest benefit.
  • CONCLUSION: Whole brain radiotherapy is not innocuous in young patients with germ cell tumors and can cause late CNS toxicity.
  • [MeSH-major] Brain / radiation effects. Brain Neoplasms / radiotherapy. Cranial Irradiation / adverse effects. Neoplasms, Germ Cell and Embryonal / radiotherapy. Radiation Injuries / complications. Testicular Neoplasms
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / adverse effects. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Choriocarcinoma / blood. Choriocarcinoma / drug therapy. Choriocarcinoma / radiotherapy. Choriocarcinoma / secondary. Chorionic Gonadotropin / blood. Cisplatin / administration & dosage. Combined Modality Therapy / adverse effects. Combined Modality Therapy / methods. Fatal Outcome. Humans. Leukoencephalopathy, Progressive Multifocal / etiology. Lung Neoplasms / secondary. Male. Neoplasm Proteins / blood. Radiotherapy Dosage. Salvage Therapy / methods. Stem Cell Transplantation. Time Factors

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  • [CommentIn] Int J Radiat Oncol Biol Phys. 2008 Apr 1;70(5):1300-2 [18374219.001]
  • (PMID = 18374223.001).
  • [ISSN] 0360-3016
  • [Journal-full-title] International journal of radiation oncology, biology, physics
  • [ISO-abbreviation] Int. J. Radiat. Oncol. Biol. Phys.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Chorionic Gonadotropin; 0 / Neoplasm Proteins; Q20Q21Q62J / Cisplatin
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18. Sakurada K, Saino M, Mouri W, Sato A, Kitanaka C, Kayama T: Nestin expression in central nervous system germ cell tumors. Neurosurg Rev; 2008 Apr;31(2):173-6; discussion 176-7

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  • [Title] Nestin expression in central nervous system germ cell tumors.
  • Central nervous system (CNS) germ cell tumors constitute a unique class of rare tumors that mainly affect children and adolescents.
  • Recently, results of treatment of germ cell tumors have improved with use of radiotherapy and combination chemotherapy.
  • However, some tumors have proven refractory to intensive treatment with surgery, radiation, and combination chemotherapy.
  • Nestin is an intermediate filament protein expressed in undifferentiated cells during CNS development and in CNS tumors and is used as a marker of immature elements of tumors, including brain tumor stem cells.
  • In this study, we examined for the first time nestin expression in 19 CNS germ cell tumors (nine pure germinomas, five germinomas with syncytiotrophoblastic giant cells, one yolk sac tumor, one choriocarcinoma, one embryonal carcinoma, and two mature teratomas).
  • These findings suggest that the detection of nestin expression could be useful in the management of CNS germ cell tumors, as an auxiliary predictor of dissemination and/or progression.
  • [MeSH-major] Biomarkers, Tumor / biosynthesis. Biomarkers, Tumor / genetics. Central Nervous System Neoplasms / genetics. Intermediate Filament Proteins / biosynthesis. Intermediate Filament Proteins / genetics. Neoplasms, Germ Cell and Embryonal / genetics. Nerve Tissue Proteins / biosynthesis. Nerve Tissue Proteins / genetics
  • [MeSH-minor] Adolescent. Adult. Child. Choriocarcinoma / genetics. Choriocarcinoma / metabolism. Choriocarcinoma / pathology. Endodermal Sinus Tumor / genetics. Endodermal Sinus Tumor / metabolism. Endodermal Sinus Tumor / pathology. Female. Germinoma / genetics. Germinoma / metabolism. Germinoma / pathology. Giant Cell Tumors / genetics. Giant Cell Tumors / metabolism. Giant Cell Tumors / pathology. Humans. Hypopituitarism / etiology. Immunoenzyme Techniques. Magnetic Resonance Imaging. Male. Nestin. Teratoma / genetics. Teratoma / metabolism. Teratoma / pathology. Vision Disorders / etiology

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  • (PMID = 18092184.001).
  • [ISSN] 0344-5607
  • [Journal-full-title] Neurosurgical review
  • [ISO-abbreviation] Neurosurg Rev
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / Intermediate Filament Proteins; 0 / NES protein, human; 0 / Nerve Tissue Proteins; 0 / Nestin
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19. Cohn DA, Stuart-Harris R: Isolated central nervous system relapse of non-seminomatous germ cell tumour of the testis. A case report and review of the literature. Oncology; 2001;61(3):184-8
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  • [Title] Isolated central nervous system relapse of non-seminomatous germ cell tumour of the testis. A case report and review of the literature.
  • Isolated central nervous system (CNS) relapse of non-seminomatous germ cell tumour (NSGCT) of the testis has been reported in only 12 patients previously.
  • We report a patient with an isolated CNS relapse of NSGCT, following a prior systemic relapse.
  • From a review of previous cases, isolated CNS relapse appears to be more common in patients with embryonal cell histology (alone or mixed with other elements) and occurred after a median of 8.5 months following initial presentation.
  • Long-term survival appears possible using multi-modal treatment with whole-brain radiotherapy, surgery and/or chemotherapy.
  • However, the optimal treatment of isolated CNS relapse remains undefined.
  • [MeSH-major] Brain Neoplasms / secondary. Carcinoma, Embryonal / secondary. Occipital Lobe. Testicular Neoplasms / pathology
  • [MeSH-minor] Adult. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Biomarkers, Tumor / analysis. Cisplatin / administration & dosage. Combined Modality Therapy. Cranial Irradiation. Etoposide / administration & dosage. Humans. Lung Neoplasms / drug therapy. Lung Neoplasms / secondary. Lymphatic Metastasis. Male. Orchiectomy. Radiotherapy, Adjuvant. Remission Induction. Tomography, X-Ray Computed. Vision Disorders / etiology. alpha-Fetoproteins / analysis

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  • [Copyright] Copyright 2001 S. Karger AG, Basel
  • (PMID = 11574772.001).
  • [ISSN] 0030-2414
  • [Journal-full-title] Oncology
  • [ISO-abbreviation] Oncology
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / alpha-Fetoproteins; 6PLQ3CP4P3 / Etoposide; Q20Q21Q62J / Cisplatin
  • [Number-of-references] 10
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20. Souweidane MM, Krieger MD, Weiner HL, Finlay JL: Surgical management of primary central nervous system germ cell tumors: proceedings from the Second International Symposium on Central Nervous System Germ Cell Tumors. J Neurosurg Pediatr; 2010 Aug;6(2):125-30
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  • [Title] Surgical management of primary central nervous system germ cell tumors: proceedings from the Second International Symposium on Central Nervous System Germ Cell Tumors.
  • The successful treatment of children with a primary CNS germ cell tumor can be greatly influenced by the neurosurgeon involved in the diagnostic and therapeutic care of these children.
  • Variability in surgical philosophies no doubt exists due to the relatively infrequent incidence of these tumors, a lack of consensus regarding diagnostic and therapeutic approaches, and the advent of recent surgical innovations.
  • Many of these issues were discussed at the Second International Symposium on Central Nervous System Germ Cell Tumors through presented abstracts and invited presentations.
  • [MeSH-major] Brain Neoplasms / surgery. Neoplasms, Germ Cell and Embryonal / surgery
  • [MeSH-minor] Biomarkers, Tumor / cerebrospinal fluid. Biopsy. Brain / pathology. Brain / surgery. Child. Combined Modality Therapy. Diagnostic Imaging. Endoscopy / methods. Female. Germinoma / drug therapy. Germinoma / pathology. Germinoma / radiotherapy. Germinoma / surgery. Humans. Male. Microsurgery. Neoadjuvant Therapy. Neoplasm Recurrence, Local / drug therapy. Neoplasm Recurrence, Local / pathology. Neoplasm Recurrence, Local / radiotherapy. Neoplasm Recurrence, Local / surgery. Neuronavigation. Reoperation

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  • (PMID = 20672932.001).
  • [ISSN] 1933-0715
  • [Journal-full-title] Journal of neurosurgery. Pediatrics
  • [ISO-abbreviation] J Neurosurg Pediatr
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Biomarkers, Tumor
  • [Number-of-references] 45
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21. Yang QY, Sun XF, Huang HQ, Zhen ZJ, Xia Y, Cai QQ, Ling JY: [Treatment outcome of primary central nervous system germ cell tumors after combined therapy: a report of 23 cases]. Ai Zheng; 2008 Apr;27(4):438-41
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  • [Title] [Treatment outcome of primary central nervous system germ cell tumors after combined therapy: a report of 23 cases].
  • BACKGROUND & OBJECTIVE: Primary central nervous system (CNS) germ cell tumors (GCTs) are rare malignant neoplasms with various histological types.
  • Excluding pure germinoma and mature teratoma, other types carry a poor prognosis.
  • Previous investigations focused on combined modality treatment including chemotherapy to improve survival.
  • This study was to analyze the efficacy and toxicity of chemotherapy combined with surgery and/or radiotherapy on CNS GCTs.
  • METHODS: A total of 23 patients with CNS GCTs were treated in Cancer Center of Sun Yat-sen University from May 2002 to Jun. 2006.
  • All patients were treated with chemotherapy of PEB regimen combined with surgery and/or radiotherapy.
  • RESULTS: Of the 23 patients, 17 newly diagnosed patients received induction chemotherapy followed by radiotherapy or surgery/radiotherapy followed by adjuvant chemotherapy; 6 recurrent patients received salvage chemotherapy, of which 3 patients with disseminated tumor received salvage chemotherapy followed by craniospinal irradiation.
  • Chemotherapy alone gained a response rate of 87.0% and a complete remission rate of 30.4%.
  • Incomplete tumor resection or biopsy was performed in 13 patients.
  • Fourteen patients (60.9%) were alive without evidence of diseases after combined modality treatment.
  • CONCLUSIONS: The combined modality treatment including PEB regimen is highly effective in treating CNS GCTs patients, especially in the patients with elevated tumor markers.
  • [MeSH-major] Central Nervous System Neoplasms / therapy. Neoplasms, Germ Cell and Embryonal / therapy
  • [MeSH-minor] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Bleomycin / therapeutic use. Cisplatin / therapeutic use. Combined Modality Therapy. Etoposide / therapeutic use. Female. Humans. Male. Survival Rate

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  • (PMID = 18423134.001).
  • [Journal-full-title] Ai zheng = Aizheng = Chinese journal of cancer
  • [ISO-abbreviation] Ai Zheng
  • [Language] chi
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] China
  • [Chemical-registry-number] 11056-06-7 / Bleomycin; 6PLQ3CP4P3 / Etoposide; Q20Q21Q62J / Cisplatin; BEP protocol
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22. Fuller C, Helton K, Dalton J, Fouladi M, Kun L, Burger P: Polar spongioblastoma of the spinal cord: a case report. Pediatr Dev Pathol; 2006 Jan-Feb;9(1):75-80
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  • "Rhythmic palisading" is a striking histologic pattern infrequently encountered in a variety of central nervous system (CNS) tumors.
  • We present the case of an infant with a large spinal cord lesion wherein all sampled tissue showed columnar arrangements of palisaded cells, typical of polar spongioblastoma.
  • The tumor was briskly proliferative, focally necrotic, and variably expressed S100, glial fibrillary acidic protein, neuron specific enolase, and p53 by immunohistochemistry.
  • Despite chemotherapy and decadron, he developed lesional necrosis and intracranial metastases and died less than 1 mo from presentation.
  • This case illustrates polar spongioblastoma as a distinctive histologic pattern that can occur in embryonal CNS tumors.
  • Discrimination of these rare aggressive lesions from other CNS tumors with focal palisaded architecture is crucial as the treatment and prognosis of the latter may differ significantly.
  • [MeSH-minor] Antineoplastic Agents, Hormonal / therapeutic use. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Biomarkers, Tumor / analysis. Cell Proliferation. Dexamethasone / therapeutic use. Fatal Outcome. Humans. Infant. Male

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  • (PMID = 16808641.001).
  • [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 / Antineoplastic Agents, Hormonal; 0 / Biomarkers, Tumor; 7S5I7G3JQL / Dexamethasone
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23. Székessy DP, Stoltenburg-Didinger G: Differentiation, proliferation and apoptosis in primary and recurrent primitive neuroectodermal tumors of childhood. Childs Nerv Syst; 2001 May;17(6):320-7
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  • Primitive neuroectodermal tumors (PNETs) of the CNS are a group of embryonal tumors composed of small undifferentiated or poorly differentiated cells.
  • All children underwent neurosurgery and chemotherapy according to the HIT and HIT-SKK protocols.
  • The proportion of apoptotic cells could not be related to the effect of therapy.
  • [MeSH-major] Apoptosis / physiology. Brain Neoplasms / pathology. Cell Differentiation / physiology. Cell Division / physiology. Neoplasm Recurrence, Local / pathology. Neuroectodermal Tumors, Primitive / pathology
  • [MeSH-minor] Adolescent. Adult. Biomarkers, Tumor / analysis. Brain / pathology. Brain / surgery. Chemotherapy, Adjuvant. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Infant. Male. Microscopy, Electron. Prognosis. Retrospective Studies

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  • (PMID = 11417411.001).
  • [ISSN] 0256-7040
  • [Journal-full-title] Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
  • [ISO-abbreviation] Childs Nerv Syst
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Biomarkers, Tumor
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24. Göbel U, Calaminus G, Schneider DT, Schmidt P, Haas RJ, MAKEI and MAHO Study Groups of the German Society of Pediatric Oncology and Hematology, and the SIOP CNS GCT Study Group: Management of germ cell tumors in children: approaches to cure. Onkologie; 2002 Feb;25(1):14-22
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  • The introduction of cisplatinum chemotherapy and current advances in the surgical treatment have resulted in a dramatic improvement of the prognosis of children with malignant germ cell tumors (GCT).
  • Cisplatinum chemotherapy generally results in sufficient systemic tumor control, but local relapses may still occur in patients who did not receive adequate local treatment.
  • Therefore, the therapeutic consideration must take into account age, primary site of the tumor, and its histology.
  • In gonadal tumors, there is a high chance of primary complete resection since these tumors tend to be encapsulated, and particularly testicular GCT are often detected at a low tumor stage.
  • In these tumors, a neoadjuvant or preoperative chemotherapy after clinical diagnosis by imaging and evaluation of tumor markers significantly facilitates complete resection on delayed surgery.
  • In addition, the impact of chemotherapy on local tumor control may be enhanced by locoregional hyperthermia.
  • In intracranial GCT, radiotherapy significantly contributes to local tumor control, and doses are stratified according to histology.
  • These general considerations have been integrated into national and international cooperative treatment protocols.
  • In most current protocols, treatment is stratified according to an initial risk assessment that includes the parameters age, site, histology, stage, completeness of resection and the tumor markers alpha(1)-fetoprotein (AFP) and human choriogonadotropin (beta-HCG).
  • Moreover, the previously high-risk groups may now expect a favorable prognosis with this risk-adapted treatment, whereas an increasing number of low-risk patients are treated expectantly or with significantly reduced chemotherapy.
  • [MeSH-major] Neoplasms, Germ Cell and Embryonal / therapy
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Chorionic Gonadotropin, beta Subunit, Human / blood. Cisplatin / administration & dosage. Cisplatin / adverse effects. Combined Modality Therapy. Female. Humans. Infant. Infant, Newborn. Male. Neoadjuvant Therapy. Neoplasm Staging. Prognosis. alpha-Fetoproteins / metabolism

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  • [Copyright] Copyright 2002 S. Karger GmbH, Freiburg
  • (PMID = 11893878.001).
  • [ISSN] 0378-584X
  • [Journal-full-title] Onkologie
  • [ISO-abbreviation] Onkologie
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Chorionic Gonadotropin, beta Subunit, Human; 0 / alpha-Fetoproteins; Q20Q21Q62J / Cisplatin
  • [Number-of-references] 66
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25. Echevarría ME, Fangusaro J, Goldman S: Pediatric central nervous system germ cell tumors: a review. Oncologist; 2008 Jun;13(6):690-9

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  • [Title] Pediatric central nervous system germ cell tumors: a review.
  • Central nervous system (CNS) germ cell tumors (GCTs) represent approximately 3% of primary pediatric brain tumors and encompass a wide pathologic spectrum.
  • CNS GCTs are most commonly located in the pineal and suprasellar regions of the brain and can be divided into major groups including germinomas and nongerminomatous GCTs (NGGCTs), with teratomas often considered a separate category.
  • The rare exceptions are the cases where characteristic elevations of tumor markers, including alpha-fetoprotein and/or beta-human chorionic gonadotropin are documented in the serum and/or cerebrospinal fluid.
  • In these cases, the imaging findings along with the tumor marker elevation may be diagnostic in themselves without the need for tissue confirmation.
  • Treatment and prognosis differ greatly between groups.
  • More recently, the use of chemotherapy in addition to radiation therapy has afforded the ability to decrease the dose and volume of radiation therapy without affecting survival rates.
  • NGGCTs are less radiosensitive than germinomas, but the use of adjuvant chemotherapy has improved survival rates in this group as well.
  • The standard management for CNS GCTs remains controversial.
  • Treatment regimens aimed to improve progression-free and overall survival times are ongoing.
  • [MeSH-major] Central Nervous System Neoplasms / pathology. Neoplasms, Germ Cell and Embryonal / pathology
  • [MeSH-minor] Child. Combined Modality Therapy. Humans

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  • (PMID = 18586924.001).
  • [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] 75
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26. Fouladi M, Blaney SM, Poussaint TY, Freeman BB 3rd, McLendon R, Fuller C, Adesina AM, Hancock ML, Danks MK, Stewart C, Boyett JM, Gajjar A: Phase II study of oxaliplatin in children with recurrent or refractory medulloblastoma, supratentorial primitive neuroectodermal tumors, and atypical teratoid rhabdoid tumors: a pediatric brain tumor consortium study. Cancer; 2006 Nov 1;107(9):2291-7
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  • [Title] Phase II study of oxaliplatin in children with recurrent or refractory medulloblastoma, supratentorial primitive neuroectodermal tumors, and atypical teratoid rhabdoid tumors: a pediatric brain tumor consortium study.
  • BACKGROUND: An open-label Phase II study of oxaliplatin was conducted to evaluate its safety and efficacy in children with recurrent or refractory medulloblastoma (MB), supratentorial primitive neuroectodermal tumors (SPNET), and atypical teratoid rhabdoid tumor (ATRT).
  • CONCLUSIONS: Oxaliplatin was well tolerated in children but has limited activity in children with recurrent CNS embryonal tumors previously treated with platinum compounds.
  • [MeSH-major] Medulloblastoma / drug therapy. Neoplasm Recurrence, Local / drug therapy. Neuroectodermal Tumors, Primitive / drug therapy. Organoplatinum Compounds / therapeutic use. Rhabdoid Tumor / drug therapy. Supratentorial Neoplasms / drug therapy. Teratoma / drug therapy
  • [MeSH-minor] Adolescent. Antineoplastic Agents / administration & dosage. Antineoplastic Agents / pharmacokinetics. Antineoplastic Agents / therapeutic use. Child. Child, Preschool. Female. Humans. Infant. Male. Treatment Outcome


27. Lee JY, Kim BS, Phi JH, Kang HJ, Park SH, Wang KC, Kim IH, Cho BK, Kim SK: Primary meningeal rhabdomyosarcoma associated with chronic subdural effusion: case report. J Neurosurg Pediatr; 2010 Feb;5(2):167-71

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  • Primary sarcomas of the CNS are rare and are sometimes associated with chronic subdural effusion (SDE).
  • When he presented with acute headache, nausea, and vomiting, a newly developed tumor was found.
  • Near-total resection of the tumor was performed, and the mass was diagnosed as an embryonal-type rhabdomyosarcoma.
  • The child was given radiation therapy and 13 cycles of chemotherapy and is still free of disease 13 months after surgery.
  • [MeSH-minor] Cerebrospinal Fluid Shunts. Child, Preschool. Chronic Disease. Craniotomy. Epidural Space / surgery. Humans. Immunohistochemistry. Male. Neurosurgical Procedures. Seizures / etiology. Tomography, X-Ray Computed

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  • (PMID = 20121365.001).
  • [ISSN] 1933-0715
  • [Journal-full-title] Journal of neurosurgery. Pediatrics
  • [ISO-abbreviation] J Neurosurg Pediatr
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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28. Bhat SR, Goodwin TL, Burwinkle TM, Lansdale MF, Dahl GV, Huhn SL, Gibbs IC, Donaldson SS, Rosenblum RK, Varni JW, Fisher PG: Profile of daily life in children with brain tumors: an assessment of health-related quality of life. J Clin Oncol; 2005 Aug 20;23(24):5493-500
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  • PURPOSE: The survival of children with CNS tumors approaches 70%, yet health-related quality of life (HRQOL) has not been investigated rigorously in this population.
  • RESULTS: One hundred thirty-four patients (73 male; mean age +/- standard deviation, 11.8 +/- 5.4 years; 55 had low-grade glioma, 32 had medulloblastoma/primitive neuroectodermal tumor/embryonal tumor, 17 had malignant astrocytoma, nine had germ-cell tumor, and 21 had other types of tumors) were assessed, each in less than 20 minutes.
  • Children receiving radiation therapy (XRT) but no chemotherapy had significantly lower total, psychosocial, emotional, and social functioning than those receiving other treatments, including XRT plus chemotherapy.
  • CONCLUSION: The PedsQL can be used to assess HRQOL rapidly and easily in children with CNS tumors, who have significantly worse HRQOL than healthy children.
  • Children receiving XRT fare worse overall; chemotherapy added to XRT does not seem to worsen HRQOL.

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  • [CommentIn] J Clin Oncol. 2005 Aug 20;23(24):5424-6 [16110000.001]
  • (PMID = 16110009.001).
  • [ISSN] 0732-183X
  • [Journal-full-title] Journal of clinical oncology : official journal of the American Society of Clinical Oncology
  • [ISO-abbreviation] J. Clin. Oncol.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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29. Frühwald MC, Rickert CH, O'Dorisio MS, Madsen M, Warmuth-Metz M, Khanna G, Paulus W, Kühl J, Jürgens H, Schneider P, Müller HL: Somatostatin receptor subtype 2 is expressed by supratentorial primitive neuroectodermal tumors of childhood and can be targeted for somatostatin receptor imaging. Clin Cancer Res; 2004 May 1;10(9):2997-3006
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  • PURPOSE: Although gliomas predominate among central nervous system (CNS) neoplasms in adulthood, embryonal tumors are the most common malignant brain tumors in children.
  • Despite novel treatment approaches, including improved radiotherapy and high-dose chemotherapy, survival rates remain unsatisfactory.
  • The timely diagnosis of residual or recurrent embryonal CNS tumors and thus the earliest possible time point for intervention is often hampered by inaccuracies of conventional imaging techniques.
  • Here, we evaluated somatostatin receptor type 2 (sst(2)) expression using an antibody in an array of CNS tumors of childhood.
  • RESULTS: Abundant expression of somatostatin receptor type 2 in stPNET, a ME, and ependymomas warranted in vivo imaging of 7 stPNET, 1 rhabdomyosarcoma, 3 ependymomas, 1 ME, and 1 glioblastoma.
  • In selected cases SRI was more sensitive in the detection of relapse than conventional imaging by magnetic resonance imaging and computed tomography.
  • CONCLUSIONS: SRI should be considered in the evaluation of residual or recurrent embryonal CNS tumors, especially stPNET.
  • The strengths of SRI lie in the differentiation of reactive tissue changes versus residual or recurrent tumor, the detection of small lesions, and possibly in the distinction of stPNET from gliomas.
  • [MeSH-minor] Central Nervous System Neoplasms / metabolism. Central Nervous System Neoplasms / pathology. Central Nervous System Neoplasms / radionuclide imaging. Child. Child, Preschool. Female. Humans. Immunohistochemistry. Infant. Male. Tomography, Emission-Computed, Single-Photon / methods

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  • (PMID = 15131035.001).
  • [ISSN] 1078-0432
  • [Journal-full-title] Clinical cancer research : an official journal of the American Association for Cancer Research
  • [ISO-abbreviation] Clin. Cancer Res.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Receptors, Somatostatin; 0 / somatostatin receptor 2
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30. Kantar M, Cetingül N, Kansoy S, Anacak Y, Demirtaş E, Erşahin Y, Mutluer S: Radiotherapy-induced secondary cranial neoplasms in children. Childs Nerv Syst; 2004 Jan;20(1):46-9
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  • BACKGROUND: Secondary malignant neoplasms (SMN) in CNS tumor survivors has become problem of increasing concern over the last 20 years.
  • These tumors usually occur in a different site from the primary brain tumor several years after treatment.
  • CASE REPORT: We report secondary cranial malignant neoplasms in three patients who were treated with irradiation and chemotherapy for their primary brain tumors.
  • The first case is a male survivor of an orbital rhabdomyosarcoma who developed a meningioma 8 years later.
  • The other two cases are female survivors of ependymomas who were irradiated at the age of 3 and developed secondary gliomas 8 and 17 years after therapy respectively.
  • CONCLUSION: Patients carry a risk of developing SMNs many years later since irradiation is still an important part of the treatment.
  • [MeSH-major] Meningioma / etiology. Radiotherapy / adverse effects. Rhabdomyosarcoma, Embryonal / etiology

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  • [Cites] Pediatr Radiol. 2001 Sep;31(9):607-9 [11511997.001]
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  • (PMID = 14714135.001).
  • [ISSN] 0256-7040
  • [Journal-full-title] Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
  • [ISO-abbreviation] Childs Nerv Syst
  • [Language] eng
  • [Publication-type] Case Reports; Comparative Study; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antibodies, Antinuclear; 0 / Antibodies, Monoclonal; 0 / Desmin; 0 / Glial Fibrillary Acidic Protein; 0 / Ki-67 Antigen; 0 / MIB-1 antibody; 0 / S100 Proteins
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31. Raney B, Anderson J, Breneman J, Donaldson SS, Huh W, Maurer H, Michalski J, Qualman S, Ullrich F, Wharam M, Meyer W, Soft-Tissue Sarcoma Committee of the Children's Oncology Group, Arcadia, California, USA: Results in patients with cranial parameningeal sarcoma and metastases (Stage 4) treated on Intergroup Rhabdomyosarcoma Study Group (IRSG) Protocols II-IV, 1978-1997: report from the Children's Oncology Group. Pediatr Blood Cancer; 2008 Jul;51(1):17-22
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Treatment included vincristine, actinomycin D, and cyclophosphamide (VAC) chemotherapy and radiotherapy to the primary tumor and up to five metastatic sites/tissues.
  • Sixty patients had progressive disease (N = 49) or death as a first event (N = 11); another developed myelodysplastic syndrome and died.
  • Sites of first progression/relapse were distant (55%), local (12%), CNS extension (8%), mixed (6%), and uncertain (18%).
  • Factors indicating likelihood of 10-year FFS included tumor arising in "better" versus "worse" sites (FFS 46% vs. 18%, P = 0.02) and embryonal versus other histology (FFS 37% vs. 19%, P = 0.06).
  • Patients with the best outlook had embryonal RMS located in the nasopharynx/nasal cavity, middle ear/mastoid, or parapharyngeal region.
  • Distant metastases were the most frequent type of recurrence, indicating that more effective systemic agents are needed to eliminate residual disease.

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  • [Copyright] (c) 2008 Wiley-Liss, Inc.
  • (PMID = 18266224.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] ENG
  • [Grant] United States / NCI NIH HHS / CA / CA-98543; United States / NCI NIH HHS / CA / CA-24507; United States / NCI NIH HHS / CA / U10 CA098413; United States / NCI NIH HHS / CA / CA-72989; United States / NCI NIH HHS / CA / U10 CA098543; United States / NCI NIH HHS / CA / CA-29511
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural
  • [Publication-country] United States
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32. Eberhart CG, Brat DJ, Cohen KJ, Burger PC: Pediatric neuroblastic brain tumors containing abundant neuropil and true rosettes. Pediatr Dev Pathol; 2000 Jul-Aug;3(4):346-52
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • We have encountered a series of seven unusual neuroblastic pediatric central nervous system (CNS) neoplasms with a unique constellation of histologic, immunohistochemical, and ultrastructural features.
  • True rosettes with well-formed central lumens often filled with granular debris were present, along with perivascular pseudorosettes and occasional Homer-Wright rosettes.
  • Immunohistochemically, the neuropil-like areas as well as the perinuclear cytoplasm of many embryonal tumor cells were positive for synaptophysin and neurofilament protein.
  • Ultrastructurally, the tumor cells showed microtubule-containing neuronal processes, some with neurosecretory granules.
  • The clinical outcome was poor, with five patients dead from their disease 5 to 14 months after initial presentation and one patient with recurrent disease 7 months after resection and chemotherapy.
  • [MeSH-minor] Apoptosis. Brain / metabolism. Brain / pathology. Child, Preschool. Female. Glial Fibrillary Acidic Protein / analysis. Humans. Infant. Magnetic Resonance Imaging. Male. Neutrophils. Rosette Formation. Treatment Outcome

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  • (PMID = 10890250.001).
  • [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; Comparative Study; Journal Article
  • [Publication-country] UNITED STATES
  • [Chemical-registry-number] 0 / Glial Fibrillary Acidic Protein
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