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1. Friedman HS, Keir ST, Houghton PJ: The emerging role of irinotecan (CPT-11) in the treatment of malignant glioma in brain tumors. Cancer; 2003 May 1;97(9 Suppl):2359-62
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  • [Title] The emerging role of irinotecan (CPT-11) in the treatment of malignant glioma in brain tumors.
  • Laboratory studies have demonstrated the activity of irinotecan in a broad panel of pediatric and adult central nervous system tumor xenografts in athymic nude mice.
  • These studies led to a Phase II trial that confirmed the activity of this agent in the treatment of recurrent malignant glioma.
  • These laboratory studies generated a recently completed Phase I trial of CPT-11 + BCNU, which now is being evaluated in a formal Phase II trial for adults with newly diagnosed or recurrent malignant glioma.
  • More recent studies have demonstrated similar interaction between CPT-11 and temozolomide and have led to a Phase I trial of these agents in the treatment of adults with malignant glioma.
  • Studies currently are addressing the role of O(6)-alkylguanine-DNA alkyltransferase (AGT) in reducing the benefits of combining CPT-11 with temozolomide and the potential therapeutic gain from utilizing an inhibitor of AGT.
  • [MeSH-major] Brain Neoplasms / drug therapy. Camptothecin / analogs & derivatives. Camptothecin / therapeutic use. Dacarbazine / analogs & derivatives. Glioma / drug therapy
  • [MeSH-minor] Adult. Animals. Antineoplastic Agents, Alkylating / administration & dosage. Antineoplastic Agents, Phytogenic / administration & dosage. Antineoplastic Agents, Phytogenic / therapeutic use. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Carmustine / administration & dosage. Clinical Trials as Topic. Humans. Neoplasm Recurrence, Local

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  • [Copyright] Copyright 2003 American Cancer Society.DOI 10.1002/cncr.11305
  • (PMID = 12712457.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Alkylating; 0 / Antineoplastic Agents, Phytogenic; 7673326042 / irinotecan; 7GR28W0FJI / Dacarbazine; 85622-93-1 / temozolomide; U68WG3173Y / Carmustine; XT3Z54Z28A / Camptothecin
  • [Number-of-references] 9
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2. Maria BL, Gupta N, Gilg AG, Abdel-Wahab M, Leonard AP, Slomiany M, Wheeler WG, Tolliver LB, Babcock MA, Lucas JT Jr, Toole BP: Targeting hyaluronan interactions in spinal cord astrocytomas and diffuse pontine gliomas. J Child Neurol; 2008 Oct;23(10):1214-20
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  • Although significant advances have been made in treating malignant pediatric central nervous system tumors such as medulloblastoma, no effective therapy exists for diffuse pontine glioma or intramedullary spinal astrocytoma.
  • Biology of these 2 tumors is poorly understood, in part because diffuse pontine gliomas are not treated surgically, and tumor specimens from intramedullary spinal astrocytomas are rare and minuscule.
  • At the 2007 Neurobiology of Disease in Children Symposium, we presented evidence that malignant glioma behaviors, including antiapoptosis, invasiveness, and treatment resistance, are enhanced by hyaluronan, an extracellular glycosaminoglycan.
  • We review the clinical course of pediatric intramedullary spinal astrocytoma and diffuse pontine glioma, and show expression of membrane proteins that interact with hyaluronan: CD44, extracellular matrix metalloproteinase inducer, and breast cancer resistance protein (BCRP/ABCG2).
  • Furthermore, we describe novel animal models of these tumors for preclinical studies.
  • These findings suggest that hyaluronan antagonism has potential therapeutic value in malignant central nervous system tumors.
  • [MeSH-major] Astrocytoma / metabolism. Brain Stem Neoplasms / metabolism. Glioma / metabolism. Hyaluronic Acid / metabolism. Spinal Cord Neoplasms / metabolism
  • [MeSH-minor] ATP Binding Cassette Transporter, Sub-Family G, Member 2. ATP-Binding Cassette Transporters / genetics. ATP-Binding Cassette Transporters / metabolism. Animals. Basigin / genetics. Basigin / metabolism. Child. Disease Models, Animal. Drug Resistance, Neoplasm / genetics. Humans. Hyaluronan Receptors / genetics. Hyaluronan Receptors / metabolism. Neoplasm Proteins / genetics. Neoplasm Proteins / metabolism. Rats

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  • (PMID = 18952588.001).
  • [ISSN] 1708-8283
  • [Journal-full-title] Journal of child neurology
  • [ISO-abbreviation] J. Child Neurol.
  • [Language] eng
  • [Grant] United States / NINDS NIH HHS / NS / R13 NS040925; United States / NCI NIH HHS / CA / R01 CA073839; United States / NINDS NIH HHS / NS / 5R13NS040925-09; United States / NCRR NIH HHS / RR / C06RR015455; United States / NCI NIH HHS / CA / R01 CA082867; United States / NCI NIH HHS / CA / CA073839; United States / NCRR NIH HHS / RR / C06 RR015455; United States / NCI NIH HHS / CA / CA082867
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / ABCG2 protein, human; 0 / ATP Binding Cassette Transporter, Sub-Family G, Member 2; 0 / ATP-Binding Cassette Transporters; 0 / BSG protein, human; 0 / Hyaluronan Receptors; 0 / Neoplasm Proteins; 136894-56-9 / Basigin; 9004-61-9 / Hyaluronic Acid
  • [Number-of-references] 23
  • [Other-IDs] NLM/ NIHMS463105; NLM/ PMC3641563
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3. Badruddoja MA, Keir ST, King I, Zeidner J, Vredenburgh JJ, Muhlbaier LH, Bigner DD, Friedman HS: Activity of VNP40101M (Cloretazine) in the treatment of CNS tumor xenografts in athymic mice. Neuro Oncol; 2007 Jul;9(3):240-4
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  • [Title] Activity of VNP40101M (Cloretazine) in the treatment of CNS tumor xenografts in athymic mice.
  • The current study was designed to assess the activity of VNP40101M administered at a dose of 18 mg/kg daily for five days against a panel of human adult and pediatric CNS tumors growing subcutaneously or intracranially in athymic nude mice.
  • The results demonstrated statistically significant (p < 0.05) growth delays of 15.0, 8.3, 51.0, 60+, 60+, and 60+ days in subcutaneous xenografts derived from childhood glioblastoma multiforme (D-456 MG), childhood ependymoma (D-528 EP and D-612 EP), childhood medulloblastoma (D-425 MED), and adult malignant glioma (D-245 MG and D-54 MG), respectively, with corresponding tumor regressions in 10 of 10, 4 of 10, 8 of 10, 9 of 10, 9 of 10, and 10 of 10 treated mice, respectively.
  • Delayed toxicity was seen more than 60 days after treatment, with 23 deaths in 100 treated animals, despite a median weight loss of only 0.06%.
  • In mice bearing intracranial D-245 MG xenografts, treatment with VNP40101M at a dose of 18 mg/kg daily for five days produced a 50% increase in median survival compared with controls.
  • Additional experiments conducted against subcutaneous D-245 MG xenografts by using reduced doses of 13.5 or 9.0 mg/kg daily for five days demonstrated tumor growth delays of 82.2 and 53.5 days, with corresponding tumor regressions in 8 of 9 and 9 of 10 treated mice, respectively (all values, p < 0.001), with one toxic death.
  • These findings suggest that VNP40101M is active in the treatment of a wide range of human central nervous system tumors and warrants translation to the clinic.

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  • (PMID = 17522334.001).
  • [ISSN] 1522-8517
  • [Journal-full-title] Neuro-oncology
  • [ISO-abbreviation] Neuro-oncology
  • [Language] ENG
  • [Grant] United States / NINDS NIH HHS / NS / P50 NS020023; United States / NINDS NIH HHS / NS / 5P50-NS20023-23
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Hydrazines; 0 / Prodrugs; 0 / Sulfonamides; 14J2G0U3NQ / laromustine
  • [Other-IDs] NLM/ PMC1907418
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4. Choi LM, Rood B, Kamani N, La Fond D, Packer RJ, Santi MR, Macdonald TJ: Feasibility of metronomic maintenance chemotherapy following high-dose chemotherapy for malignant central nervous system tumors. Pediatr Blood Cancer; 2008 May;50(5):970-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Feasibility of metronomic maintenance chemotherapy following high-dose chemotherapy for malignant central nervous system tumors.
  • BACKGROUND: Children less than 5 years of age with malignant central nervous system (CNS) tumors, continue to have a high rate of morbidity and mortality following administration of conventional therapy.
  • In an attempt to avoid the neurologic sequelae associated with craniospinal radiation, strategies such as high-dose chemotherapy (HDCT) followed by peripheral stem cell rescue have been used successfully.
  • Metronomic chemotherapy has also been reported as a potential new treatment strategy in solid tumors, particularly in adults.
  • PROCEDURE: A retrospective chart analysis was performed on 10 patients less than 5 years of age with CNS tumors treated with metronomic chemotherapy shortly after HDCT as part of their clinical care.
  • RESULTS: Metronomic chemotherapy was associated with minimal toxicity and all patients maintained a good quality of life.
  • At the time of this report, all 10 patients are alive.
  • Two patients have relapsed, and the remaining eight, including six patients with metastatic disease, continue to have stable clinical and radiographic disease at a mean of 20 months from the time of diagnosis.
  • CONCLUSIONS: Metronomic chemotherapy in this patient population is feasible and shows encouraging preliminary results, especially in patients with metastatic disease who have not received craniospinal radiation.
  • Further investigation of this strategy in newly diagnosed patients with CNS tumors is warranted.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / drug therapy. Ganglioglioma / drug therapy. Medulloblastoma / drug therapy. Neuroectodermal Tumors, Primitive / drug therapy. Oligodendroglioma / drug therapy
  • [MeSH-minor] Carboplatin / administration & dosage. Chemotherapy, Adjuvant. Child, Preschool. Cisplatin / administration & dosage. Cyclophosphamide / administration & dosage. Etoposide / administration & dosage. Feasibility Studies. Humans. Infant. Neoplasm Recurrence, Local / drug therapy. Neoplasm Recurrence, Local / radiotherapy. Quality of Life. Retrospective Studies. Thiotepa / administration & dosage. Treatment Outcome. Vincristine / administration & dosage

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  • [Copyright] (c) 2008 Wiley-Liss, Inc.
  • (PMID = 17941070.001).
  • [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] 5J49Q6B70F / Vincristine; 6PLQ3CP4P3 / Etoposide; 8N3DW7272P / Cyclophosphamide; 905Z5W3GKH / Thiotepa; BG3F62OND5 / Carboplatin; Q20Q21Q62J / Cisplatin
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5. Orsey A, Paessler M, Lange BJ, Nichols KE: Central nervous system juvenile xanthogranuloma with malignant transformation. Pediatr Blood Cancer; 2008 Apr;50(4):927-30
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Central nervous system juvenile xanthogranuloma with malignant transformation.
  • Here, we describe a patient with JXG diffusely involving the central nervous system (CNS), whose disease responded to therapy but subsequently underwent dissemination to the peritoneum and bone marrow.
  • Repeat biopsy at dissemination revealed pleomorphic histiocytes with tetraploidy, suggesting evolution to a clonal histiocytic neoplasm.
  • Despite further chemotherapy, the patient died of disease progression.
  • This case highlights the clinical and pathological heterogeneity of JXG and the difficulty of treating multi-focal CNS disease.
  • [MeSH-major] Brain Neoplasms / physiopathology. Brain Neoplasms / therapy. Histiocytic Sarcoma / pathology. Xanthogranuloma, Juvenile / physiopathology. Xanthogranuloma, Juvenile / therapy
  • [MeSH-minor] Anti-Inflammatory Agents / therapeutic use. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Diseases / pathology. Child. Cladribine / therapeutic use. Dexamethasone / therapeutic use. Diagnosis, Differential. Disease Progression. Fatal Outcome. Humans. Immunohistochemistry. In Situ Hybridization, Fluorescence. Magnetic Resonance Imaging. Male. Pseudotumor Cerebri / pathology. Radiotherapy


6. Norris LS, Snodgrass S, Miller DC, Wisoff J, Garvin J, Rorke LB, Finlay JL: Recurrent central nervous system medulloepithelioma: response and outcome following marrow-ablative chemotherapy with stem cell rescue. J Pediatr Hematol Oncol; 2005 May;27(5):264-6
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  • [Title] Recurrent central nervous system medulloepithelioma: response and outcome following marrow-ablative chemotherapy with stem cell rescue.
  • Medulloepithelioma is a rare primitive neuroectodermal tumor of the central nervous system usually developing in childhood, displaying highly malignant behavior, with early progression or recurrence.
  • The purpose of this study was to evaluate the efficacy of high-dose, marrow-ablative chemotherapy with autologous hemopoietic stem cell rescue in the treatment of recurrent central nervous system medulloepithelioma.
  • Three young children with recurrent central nervous system medulloepithelioma received high-dose marrow-ablative chemotherapy with thiotepa and etoposide either alone (one patient) or with the addition of carboplatin (two patients).
  • One child with residual radiographic tumor at the time of treatment could be evaluated for response and showed complete resolution of leptomeningeal disease after receiving marrow-ablative chemotherapy.
  • Two children developed tumor recurrence at 2.0 and 5.5 months after receiving marrow-ablative chemotherapy.
  • The third child continues free of tumor beyond 12 years from treatment.
  • The authors' experience with marrow-ablative chemotherapy and autologous hemopoietic stem cell rescue suggests that this treatment strategy might be beneficially incorporated into the initial treatment approach for young children with medulloepithelioma.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Carcinoma / therapy. Central Nervous System Neoplasms / therapy. Neoplasm Recurrence, Local. Stem Cell Transplantation
  • [MeSH-minor] Child, Preschool. Combined Modality Therapy. Female. Humans. Magnetic Resonance Imaging. Male. Time Factors. Treatment Outcome

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  • (PMID = 15891561.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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7. Grodman H, Wolfe L, Kretschmar C: Outcome of patients with recurrent medulloblastoma or central nervous system germinoma treated with low dose continuous intravenous etoposide along with dose-intensive chemotherapy followed by autologous hematopoietic stem cell rescue. Pediatr Blood Cancer; 2009 Jul;53(1):33-6
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  • [Title] Outcome of patients with recurrent medulloblastoma or central nervous system germinoma treated with low dose continuous intravenous etoposide along with dose-intensive chemotherapy followed by autologous hematopoietic stem cell rescue.
  • BACKGROUND: Adults and children with recurrent malignant central nervous system (CNS) tumors have a poor prognosis despite high dose chemotherapy with a conventional stem cell rescue regimen.
  • In this study we evaluated the results of low dose, continuous infusion etoposide over 21 days added to a conventional high-dose regimen of carboplatin and thiotepa in eight patients with relapsed pediatric CNS tumors.
  • PROCEDURE: Patients with high risk CNS tumors were treated with etoposide 25 mg/m(2)/day by continuous intravenous (IV) infusion from day -22 to day -2, carboplatin 667 mg/m(2)/dose IV (or area under the curve = 9 mg/ml/min according to the Calvert formula on days -8, -7, and -6, and thiotepa 300 mg/m(2)/dose IV on days -5, -4, and -3, followed by autologous hematopoietic stem cell rescue on day 0.
  • RESULTS: Eight adults and children, with a mean age of 12.9 years (age range 5.6-27.8 years), with relapsed primary CNS tumors (metastatic medulloblastoma (7), germinoma (1)), were enrolled.
  • This treatment strategy deserves further evaluation in a larger group of high-risk or relapsed primary CNS tumors.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Central Nervous System Neoplasms / therapy. Etoposide / administration & dosage. Germinoma / therapy. Hematopoietic Stem Cell Transplantation. Neoplasm Recurrence, Local / therapy
  • [MeSH-minor] Adolescent. Adult. Anemia / chemically induced. Anemia / therapy. Carboplatin / administration & dosage. Child. Child, Preschool. Female. Hematologic Diseases / chemically induced. Hematologic Diseases / therapy. Humans. Infusions, Intravenous. Male. Survival Rate. Thiotepa / administration & dosage. Treatment Outcome

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  • [Copyright] Copyright 2009 Wiley-Liss, Inc.
  • (PMID = 19326417.001).
  • [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] 6PLQ3CP4P3 / Etoposide; 905Z5W3GKH / Thiotepa; BG3F62OND5 / Carboplatin
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8. Bacci G, Ferrari C, Longhi A, Ferrari S, Forni C, Bacchini P, Palmerini E, Briccoli A, Pignotti E, Balladelli A, Picci P: Second malignant neoplasm in patients with osteosarcoma of the extremities treated with adjuvant and neoadjuvant chemotherapy. J Pediatr Hematol Oncol; 2006 Dec;28(12):774-80
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  • [Title] Second malignant neoplasm in patients with osteosarcoma of the extremities treated with adjuvant and neoadjuvant chemotherapy.
  • We evaluated the rate of second malignancies in 1205 patients with osteosarcoma of the extremity treated at our Institution with different protocols of adjuvant and neoadjuvant chemotherapy.
  • Twenty-six patients (2.15%) developed a second malignant neoplasm at a median of 7.6 years (1 to 25 y) after primary osteosarcoma treatment.
  • Of these, 2 developed a third cancer which were not considered in the series.
  • Second neoplasms were leukemia (10), breast (7), lung (2), kidney (2), central nervous system cancer (2), soft tissue (1), parotid (1), and colon (1).
  • The rate of second neoplasms was significantly higher in female patients, and the latent period shorter in hematologic tumors compared with solid tumors.
  • Ten of these 26 patients are disease free at a median of 7.7 years (range 1 to 15 y) after the last treatment.
  • The rate of second malignancies observed in the osteosarcoma group was significantly higher than that observed in the control group of 1160 patients with benign tumors treated in the same period at our Institute (2.2% vs. 0.8%, P<0.009).
  • Our study showed that the risk of second neoplasm within 15 years increased and then leveled off and that although secondary solid tumors could be explained as unrelated cases, leukemias seem to be over represented.
  • [MeSH-major] Neoplasms, Second Primary / epidemiology. Osteosarcoma
  • [MeSH-minor] Adolescent. Adult. Chemotherapy, Adjuvant / methods. Child. Child, Preschool. Disease-Free Survival. Female. Humans. Incidence. Infant. Male. Neoplasms / drug therapy. Neoplasms / epidemiology. Retrospective Studies. Risk Factors. Sex Factors. Time Factors

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  • (PMID = 17164644.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Publication-type] Comparative Study; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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9. Zimmerman MA, Goumnerova LC, Proctor M, Scott RM, Marcus K, Pomeroy SL, Turner CD, Chi SN, Chordas C, Kieran MW: Continuous remission of newly diagnosed and relapsed central nervous system atypical teratoid/rhabdoid tumor. J Neurooncol; 2005 Mar;72(1):77-84
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Continuous remission of newly diagnosed and relapsed central nervous system atypical teratoid/rhabdoid tumor.
  • Atypical teratoid/rhabdoid tumors (AT/RT) are highly malignant lesions of childhood that carry a very poor prognosis.
  • AT/RT can occur in the central nervous system (CNS AT/RT) and disease in this location carries an even worse prognosis with a median survival of 7 months.
  • In spite of multiple treatment regimens consisting of maximal surgical resection (including second look surgery), radiation therapy (focal and craniospinal), and multi-agent intravenous, oral and intrathecal chemotherapy, with or without high-dose therapy and stem cell rescue, only seven long-term survivors of CNS AT/RT have been reported, all in patients with newly diagnosed disease.
  • For this reason, many centers now direct such patients, particularly those under 5 years of age, or those with recurrent disease, towards comfort care rather than attempt curative therapy.
  • We now report on four children, two with newly diagnosed CNS AT/RT and two with progressive disease after multi-agent chemotherapy who are long term survivors (median follow-up of 37 months) using a combination of surgery, radiation therapy, and intensive chemotherapy.
  • The chemotherapy component was modified from the Intergroup Rhabdomyosarcoma Study Group (IRS III) parameningeal protocol as three of the seven reported survivors in the literature were treated using this type of therapy.
  • Our four patients, when added to the three reported survivors in the literature using this approach, suggest that patients provided this aggressive therapy can significantly alter the course of their disease.
  • More importantly, we report on the first two survivors after relapse with multi-agent intravenous and intrathecal chemotherapy treated with this modified regimen.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / therapy. Neoplasm Recurrence, Local / therapy. Rhabdoid Tumor / therapy. Teratoma / therapy
  • [MeSH-minor] Chemotherapy, Adjuvant / methods. Child. Child, Preschool. Female. Humans. Infant. Male. Radiotherapy, Adjuvant / methods. Remission Induction. Survival Analysis

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  • (PMID = 15803379.001).
  • [ISSN] 0167-594X
  • [Journal-full-title] Journal of neuro-oncology
  • [ISO-abbreviation] J. Neurooncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] United States
  • [Number-of-references] 37
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10. Roland JT Jr, Cosetti M, Liebman T, Waltzman S, Allen JC: Cochlear implantation following treatment for medulloblastoma. Laryngoscope; 2010 Jan;120(1):139-43
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  • [Title] Cochlear implantation following treatment for medulloblastoma.
  • OBJECTIVES/HYPOTHESIS: Medulloblastoma is the most common pediatric malignant tumor of the central nervous system in children.
  • Treatment includes surgical excision, external beam radiation, and multiagent chemotherapy.
  • Otologic sequelae are common and may result from radiation and/or chemotherapy.
  • Profound sensorineural hearing loss (SNHL) is a known complication of neuro-oncologic treatment and may render these patients eligible for cochlear implantation (CI).
  • Issues of CI in this population, including diagnosis, treatment of preoperative middle ear disease, operative and postoperative course, performance data, and long-term tumor surveillance are highlighted and reviewed.
  • METHODS: Three patients treated for pediatric medulloblastoma with surgical resection, postoperative hyperfractioned craniospinal radiotherapy, and multiagent adjuvant chemotherapy who underwent cochlear implantation were identified.
  • Details of neuro-oncologic treatment and associated otologic complications are presented and analyzed.
  • Primary outcome assessment includes treatment of middle ear pathology, perioperative cochlear implant course, and postimplantation performance data.
  • RESULTS: Each patient required surgical treatment of chronic ear disease 4 to 16 years after chemoradiation.
  • All progressed to profound SNHL and were implanted 8 to 17 years post-neuro-oncologic treatment.
  • One patient developed postoperative wound dehiscence requiring operative closure.
  • CONCLUSIONS: Patients treated for pediatric medulloblastoma develop otologic sequelae, including profound SNHL, and may require cochlear implantation.
  • [MeSH-major] Brain Neoplasms / therapy. Cochlear Implantation. Hearing Loss, Sensorineural / surgery. Medulloblastoma / therapy
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Combined Modality Therapy. Humans. Retrospective Studies. Treatment Outcome


11. Johnston DL, Keene D, Bartels U, Carret AS, Crooks B, Eisenstat D, Fryer C, Lafay-Cousin L, Larouche V, Moghrabi A, Wilson B, Zelcer S, Silva M, Brossard J, Bouffet E: Patterns of enrollment of infants with central nervous system tumours on cooperative group studies: a report from the Canadian Pediatric Brain Tumour Consortium. J Neurooncol; 2010 Sep;99(2):243-9
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  • [Title] Patterns of enrollment of infants with central nervous system tumours on cooperative group studies: a report from the Canadian Pediatric Brain Tumour Consortium.
  • In children under the age of 3, the most common solid tumours are brain tumors.
  • Treatment for many of these patients includes surgery, chemotherapy and rarely radiation therapy.
  • Many clinical trials have been performed in an attempt to establish the best treatment for these patients.
  • Patients enrolled on clinical trials contribute to the establishment of the best therapy.
  • A data bank was established using data collected from Canadian pediatric oncology centers on children less than age 3 diagnosed with brain tumours between 1990 and 2005.
  • Data were collected on the use of adjunctive treatment after surgery, treatment on a protocol, reasons patients were not registered on a protocol, and reasons for discontinuation of therapy.
  • From the 579 cases in the data bank, 302 (52%) patients were treated with further therapy after surgery.
  • The use of further therapy after surgery was significantly higher in patients with cerebellar and brain stem tumors, patients who were over 1 year of age, patients with ependymal and embryonal tumors, and patients with high grade malignant tumors.
  • Only 62 (21%) patients were enrolled on a protocol for therapy.
  • The therapy was stopped because of completion of the protocol in 50% and because of disease progression in 34%.
  • In Canada, about half of children under the age of 36 months with brain tumors are undergoing therapy following surgery for their malignancy but only a small fraction of them are enrolled on a clinical trial.
  • There needs to be improved availability of clinical trials for these patients so that novel therapies can be evaluated and survival improved.
  • [MeSH-major] Brain Neoplasms / epidemiology. Child Health Services / utilization. Glioma / epidemiology. Patient Participation. Patient Selection
  • [MeSH-minor] Canada / epidemiology. Child, Preschool. Combined Modality Therapy. Female. Humans. Infant. Infant, Newborn. Male. Neoplasm Staging. Survival Rate. Treatment Outcome

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  • (PMID = 20135195.001).
  • [ISSN] 1573-7373
  • [Journal-full-title] Journal of neuro-oncology
  • [ISO-abbreviation] J. Neurooncol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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12. Valera ET, Lucio-Eterovic AK, Neder L, Scrideli CA, Machado HR, Carlotti-Junior CG, Queiroz RG, Motta FJ, Tone LG: Quantitative PCR analysis of the expression profile of genes related to multiple drug resistance in tumors of the central nervous system. J Neurooncol; 2007 Oct;85(1):1-10
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  • [Title] Quantitative PCR analysis of the expression profile of genes related to multiple drug resistance in tumors of the central nervous system.
  • OBJECTIVES: To evaluate and compare the profile of expression of genes related to drug resistance in brain tumors and to analyze the impact of the increased expression of these genes on overall survival.
  • METHODS: Eighty microdissected brain tumor samples from 79 patients were analyzed by RQ-PCR for the genes MDR1, MRP1, MRP3, LRP and BCRP.
  • Pediatric cases (0 to 20 years): 46 (17F:29M, median age 7.3 +/- 5.9 years); adult tumors: 33 (17F:16M, median age 46.6 +/- 14.5 years).
  • RESULTS: glial tumors expressed higher MDR1 (P = 0.003) and BCRP (P = 0.03) levels than embryonic tumors.
  • Increased expression of resistance genes, separately or jointly, was not correlated with shorter overall survival in patients with medulloblastomas/PNET and malignant gliomas.
  • CONCLUSIONS: Drug resistance genes do not explain the higher sensitivity of gliomas of the hypothalamus/pituitary/optic pathways to chemotherapy.
  • High MDR1, MRP3 and LRP levels may be implicated in the primary resistance of pilocytic astrocytomas to chemotherapy.
  • [MeSH-major] Central Nervous System Neoplasms / genetics. Drug Resistance, Multiple / genetics. Drug Resistance, Neoplasm / genetics. Gene Expression Regulation, Neoplastic / genetics. Gene Expression Regulation, Neoplastic / physiology. Reverse Transcriptase Polymerase Chain Reaction / methods
  • [MeSH-minor] Adolescent. Adult. Aged. Brain Neoplasms / drug therapy. Brain Neoplasms / genetics. Brain Neoplasms / mortality. Calibration. Child. Child, Preschool. DNA Primers. Female. Gene Expression Profiling. Glioma / drug therapy. Glioma / genetics. Glioma / mortality. Humans. Immunohistochemistry. Infant. Male. Medulloblastoma / drug therapy. Medulloblastoma / genetics. Medulloblastoma / mortality. Middle Aged. RNA, Neoplasm / biosynthesis. RNA, Neoplasm / genetics. Survival Analysis

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  • (PMID = 17429576.001).
  • [ISSN] 0167-594X
  • [Journal-full-title] Journal of neuro-oncology
  • [ISO-abbreviation] J. Neurooncol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / DNA Primers; 0 / RNA, Neoplasm
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13. Morgenstern DA, Gibson S, Brown T, Sebire NJ, Anderson J: Clinical and pathological features of paediatric malignant rhabdoid tumours. Pediatr Blood Cancer; 2010 Jan;54(1):29-34
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  • [Title] Clinical and pathological features of paediatric malignant rhabdoid tumours.
  • BACKGROUND: Malignant rhabdoid tumours (MRT) and their central nervous system (CNS) counterparts atypical teratoid/rhabdoid tumours (ATRT) are rare, highly aggressive malignant neoplasms of childhood.
  • Although there are isolated reports of long-term survival with intensive, multimodal therapy, outcomes are generally poor.
  • PROCEDURE: We conducted a retrospective review of all patients diagnosed with MRT/ATRT at Great Ormond Street Hospital over the 20 years from 1989 to 2009.
  • There were four long-term survivors (>30 months), all of whom received chemotherapy with or without surgical resection or radiotherapy.
  • CONCLUSIONS: In view of poor outcomes, there is a clear need for new treatment strategies and the identification of novel molecular targets for MRT/ATRT.
  • [MeSH-major] Rhabdoid Tumor / pathology. Teratoma / pathology
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Biomarkers, Tumor / analysis. Child. Combined Modality Therapy. Female. Humans. Immunoenzyme Techniques. Infant. Male. Neoplasm Staging. Prognosis. Radiotherapy. Retrospective Studies. Survival Rate. Treatment Outcome

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  • [Copyright] Copyright 2009 Wiley-Liss, Inc.
  • (PMID = 19653294.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Biomarkers, Tumor
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14. Popadiuk S, Korzon M, Chybicka A, Szmyd K, Balwierz W, Trelinska J, Kowalczyk J, Wisniewska-Slusarz H, Woźniak W, Bilska K, Wachowiak J, Wysocki M, Krawczuk-Rybak M, Szumera M, Sznurkowska K, Renke J: [Analysis of risk factor treatment failures in therapeutic programme for malignant germ cell tumours in children. Multicentre prospective study of Polish Pediatric Group for Solid Tumours 1998--2006]. Med Wieku Rozwoj; 2007 Jul-Sep;11(3 Pt 2):301-6

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  • [Title] [Analysis of risk factor treatment failures in therapeutic programme for malignant germ cell tumours in children. Multicentre prospective study of Polish Pediatric Group for Solid Tumours 1998--2006].
  • AIMS: The aim of the study was the analysis of risk factors of therapeutic failures in children with malignant germ cell tumours treated within the multicentre programme of PPGGL from 1999--2006.
  • MATERIALS AND METHODS: The investigated group included 18 (14.3%) patients, of 123 who have finished the treatment of malignant germ cell tumour, in whom no remission was obtained or relapse occurred.
  • RESULTS: Among 18 patients with therapeutic failures 12 died.
  • Two patients from the high risk group died of complications of the treatment--sepsis during neutropenia after chemotherapy and one after haemorrhage to the central nervous system.
  • All the patients are alive in remission after second line therapy, with 78 months (median) of follow-up.
  • The main risk factor for therapeutic failures in malignant germ cell tumours was primary chemoresistance in inoperable tumours of the sacrococcygeal region.
  • 2. The mortality of treatment complications was low.
  • 3. The relapse of cancer was not a risk factor for therapeutic failure due to the high probability of second remission 4.
  • Therapeutic failures are mainly observed in patients with mixed germ cell tumour with components of yolk sac tumour or carcinoma embrionale.
  • [MeSH-major] Neoplasm Recurrence, Local. Neoplasms, Germ Cell and Embryonal / therapy
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Drug Resistance, Neoplasm. Female. Humans. Infant. Male. Poland. Risk Factors. Treatment Failure

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  • (PMID = 18663271.001).
  • [Journal-full-title] Medycyna wieku rozwojowego
  • [ISO-abbreviation] Med Wieku Rozwoj
  • [Language] pol
  • [Publication-type] Clinical Trial; English Abstract; Journal Article; Multicenter Study
  • [Publication-country] Poland
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15. Keir ST, Hausheer F, Lawless AA, Bigner DD, Friedman HS: Therapeutic activity of 7-[(2-trimethylsilyl)ethyl)]-20 (S)-camptothecin against central nervous system tumor-derived xenografts in athymic mice. Cancer Chemother Pharmacol; 2001 Jul;48(1):83-7
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  • [Title] Therapeutic activity of 7-[(2-trimethylsilyl)ethyl)]-20 (S)-camptothecin against central nervous system tumor-derived xenografts in athymic mice.
  • PURPOSE: Camptothecins have emerged as an important new class of antitumor drugs.
  • Camptothecin derivatives such as CPT-11 and topotecan are commercially available and approved for the treatment of colorectal (CPT-11) and ovarian and small-cell lung cancer (topotecan).
  • This study was designed to test the efficacy of karenitecin, a novel highly lipophilic camptothecin derivative, against a panel of human tumor xenografts derived from adult and pediatric central nervous system malignancies growing in athymic nude mice.
  • METHODS: Xenografts evaluated were derived from childhood high-grade gliomas (D-212 MG, D-456 MG), adult high-grade gliomas (D-54 MG, D-245 MG), medulloblastomas (D-341 MED, D-487 MED), and ependymomas (D-528 EP, D-612 EP), as well as sublines with demonstrated resistance to procarbazine (D-245 MG (PR)) and busulfan (D-456 (BR)).
  • CONCLUSION: These preclinical studies confirm that karenitecin is active against human central nervous system xenografts and should undergo clinical evaluation in patients with malignant central nervous system tumors.
  • [MeSH-major] Antineoplastic Agents, Phytogenic / therapeutic use. Brain Neoplasms / drug therapy
  • [MeSH-minor] Animals. Camptothecin / analogs & derivatives. Camptothecin / pharmacology. Camptothecin / therapeutic use. Female. Humans. Male. Mice. Mice, Inbred BALB C. Mice, Nude. Neoplasm Transplantation. Transplantation, Heterologous

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  • (PMID = 11488529.001).
  • [ISSN] 0344-5704
  • [Journal-full-title] Cancer chemotherapy and pharmacology
  • [ISO-abbreviation] Cancer Chemother. Pharmacol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Phytogenic; 24R60NVC41 / cositecan; 7673326042 / irinotecan; XT3Z54Z28A / Camptothecin
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16. Cavazos CM, Keir ST, Yoshinari T, Bigner DD, Friedman HS: Therapeutic activity of the topoisomerase I inhibitor J-107088 [6-N-(1-hydroxymethyla-2-hydroxyl) ethylamino-12,13-dihydro-13-(beta-D-glucopyranosyl) -5H-indolo[2,3-a]-pyrrolo[3,4-c]-carbazole-5,7(6H)-dione]] against pediatric and adult central nervous system tumor xenografts. Cancer Chemother Pharmacol; 2001 Sep;48(3):250-4
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  • [Title] Therapeutic activity of the topoisomerase I inhibitor J-107088 [6-N-(1-hydroxymethyla-2-hydroxyl) ethylamino-12,13-dihydro-13-(beta-D-glucopyranosyl) -5H-indolo[2,3-a]-pyrrolo[3,4-c]-carbazole-5,7(6H)-dione]] against pediatric and adult central nervous system tumor xenografts.
  • PURPOSE: The in vivo antitumor activity of a novel topoisomerase I inhibitor, J-107088, was tested in athymic nude mice bearing subcutaneous or intracranial pediatric and adult malignant CNS tumor-derived xenografts.
  • The xenografts evaluated were derived from a childhood glioblastoma multiforme (D-456 MG), a childhood medulloblastoma (D-341 MED), an adult anaplastic astrocytoma (D-54 MG), an adult glioblastoma multiforme (D-245 MG), and a procarbazine-resistant subline of D-245 MG [D-245 MG (PR)].
  • Growth delays ranged from 7.6 days with D-245 MG to 62.1 days with D-456 MG (P < 0.001).
  • CONCLUSION: These results indicate that J-107088 may be active in the treatment of childhood and adult malignant brain tumors and provide the rationale for initiation of clinical trials with this agent.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Brain Neoplasms / drug therapy. Carbazoles / therapeutic use. Enzyme Inhibitors / therapeutic use. Glioma / drug therapy. Glucosides / therapeutic use. Indoles. Topoisomerase I Inhibitors
  • [MeSH-minor] Adult. Animals. Child. Female. Humans. Injections, Intraperitoneal. Injections, Subcutaneous. Male. Mice. Mice, Nude. Neoplasm Transplantation. Survival Rate. Transplantation, Heterologous. Treatment Outcome. Tumor Cells, Cultured

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  • (PMID = 11592348.001).
  • [ISSN] 0344-5704
  • [Journal-full-title] Cancer chemotherapy and pharmacology
  • [ISO-abbreviation] Cancer Chemother. Pharmacol.
  • [Language] eng
  • [Grant] United States / NINDS NIH HHS / NS / 2RO1-NS30245-12; United States / NINDS NIH HHS / NS / 5P50-NS-20023-17; United States / NCI NIH HHS / CA / CA11898; etc
  • [Publication-type] Comparative Study; Journal Article; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Carbazoles; 0 / Enzyme Inhibitors; 0 / Glucosides; 0 / Indoles; 0 / Topoisomerase I Inhibitors; 1V8X590XDP / edotecarin
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17. Hurwitz CA, Strauss LC, Kepner J, Kretschmar C, Harris MB, Friedman H, Kun L, Kadota R: Paclitaxel for the treatment of progressive or recurrent childhood brain tumors: a pediatric oncology phase II study. J Pediatr Hematol Oncol; 2001 Jun-Jul;23(5):277-81
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  • [Title] Paclitaxel for the treatment of progressive or recurrent childhood brain tumors: a pediatric oncology phase II study.
  • PURPOSE: To assess the efficacy and define the toxicity of paclitaxel given at a dosage of 350 mg/m2 every 3 weeks as a 24-hour continuous infusion to children with recurrent or progressive primary brain tumors.
  • PATIENTS AND METHODS: Seventy-three eligible patients, ages 4 months to 19 years, with progressive or recurrent primary brain tumors were treated according to a Pediatric Oncology Group (POG) phase II protocol with paclitaxel (POG 9330).
  • Tumor histologic strata included: astrocytoma (n = 4), malignant glioma (n = 13), medulloblastoma (n = 16), brain stem glioma (n = 15), ependymoma (n = 13), and miscellaneous histologies (n = 12).
  • All patients had previous histologic confirmation of a primary intracranial or spinal cord tumor with magnetic resonance imaging or computed tomography documentation of unequivocally measurable progressive or recurrent disease.
  • All patients had received previous therapy including surgery, radiation therapy, and/or chemotherapy, but no patient had been previously treated on more than one phase II trial.
  • Patients were allowed to continue therapy for a total of 18 cycles in the absence of progressive disease or unacceptable toxicity.
  • RESULTS: Seventy-five patients were enrolled onto the POG 9330 protocol; two ineligible patients were removed from the study before receiving any therapy.
  • Toxicities included mild nausea, central nervous system toxicity, myelosuppression, and febrile neutropenia, including one septic death.
  • CONCLUSION: Paclitaxel is well tolerated in children with recurrent or progressive brain tumors at this dosage and schedule and may result in short-term disease stabilization in this patient population.
  • [MeSH-major] Antineoplastic Agents, Phytogenic / therapeutic use. Brain Neoplasms / drug therapy. Paclitaxel / therapeutic use
  • [MeSH-minor] Adolescent. Astrocytoma / drug therapy. Astrocytoma / pathology. Child. Child, Preschool. Dexamethasone / therapeutic use. Disease Progression. Drug Hypersensitivity / prevention & control. Ependymoma / drug therapy. Ependymoma / pathology. Female. Glioma / drug therapy. Glioma / pathology. Humans. Immunosuppressive Agents / therapeutic use. Infant. Infratentorial Neoplasms / drug therapy. Infratentorial Neoplasms / pathology. Infusions, Intravenous. Male. Medulloblastoma / drug therapy. Medulloblastoma / pathology. Nausea / chemically induced. Neoplasm Recurrence, Local. Neutropenia / chemically induced. Remission Induction. Salvage Therapy. Treatment Failure

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  • (PMID = 11464982.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA03161; United States / NCI NIH HHS / CA / CA07431; United States / NCI NIH HHS / CA / CA15525; etc
  • [Publication-type] Clinical Trial; Clinical Trial, Phase II; Journal Article; Multicenter Study; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Phytogenic; 0 / Immunosuppressive Agents; 7S5I7G3JQL / Dexamethasone; P88XT4IS4D / Paclitaxel
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18. 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-major] Brain Neoplasms / pathology. Neuroectodermal Tumors, Primitive / pathology. Neuropil / pathology
  • [MeSH-minor] Antineoplastic Agents / therapeutic use. Combined Modality Therapy. Fatal Outcome. Female. Humans. Infant. Neurosurgical Procedures. Radiotherapy

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  • [Cites] Am J Surg Pathol. 2009 Feb;33(2):211-7 [18987548.001]
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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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19. Indolfi P, Casale F, Carli M, Bisogno G, Ninfo V, Cecchetto G, Bagnulo S, Santoro N, Giuliano M, Di Tullio MT: Pleuropulmonary blastoma: management and prognosis of 11 cases. Cancer; 2000 Sep 15;89(6):1396-401
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  • BACKGROUND: Pleuropulmonary blastoma (PPB) is a rare and aggressive malignant tumor that affects children and adults.
  • This neoplasm is histologically characterized by primitive blastema and a malignant mesenchymal stroma that often demonstrates multidirectional differentiation.
  • Despite the introduction of multimodal therapy, the prognosis of patients with PPB remains poor.
  • METHODS: In the current study the authors reported on PPB cases from a national retrospective search performed in 18 Italian Associations for Pediatric Hematology and Oncology centers.
  • Clinical data, surgical notes, pathologic findings, and summaries of chemotherapy and radiotherapy were obtained from reports and correlated with outcome by standard statistical methods.
  • In three patients the PPB developed from other primary dysplastic diseases: cystic adenomatoid malformation in one case and congenital lung cysts in the other two cases.
  • Five patients experienced disease recurrences (local recurrence in three patients and distant metastasis in two patients, within the central nervous system and an intraocular location, respectively).
  • Patients with a type 2 histologic pattern and/or pleural involvement were found to have a worse outcome compared with patients without such features.
  • Event free survival at 2 years from the time of diagnosis was 45% for all patients.
  • CONCLUSIONS: PPB is an aggressive neoplasm of early childhood and to the authors' knowledge no adequate therapy has been defined to date for patients with PPB.
  • After making the diagnosis, the main goal of therapy should be radical surgery, even in patients with microscopic residual disease.
  • Because the response to chemotherapy is poor, the authors' experience suggests that chemotherapy should be given with local radiotherapy in the majority of patients.
  • [MeSH-major] Lung Neoplasms / therapy. Pleural Neoplasms / therapy. Pulmonary Blastoma / therapy
  • [MeSH-minor] Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Infant. Male. Prognosis. Survival Analysis. Treatment Outcome

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  • [Copyright] Copyright 2000 American Cancer Society.
  • (PMID = 11002236.001).
  • [ISSN] 0008-543X
  • [Journal-full-title] Cancer
  • [ISO-abbreviation] Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] UNITED STATES
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20. Koeller KK, Rushing EJ: From the archives of the AFIP: medulloblastoma: a comprehensive review with radiologic-pathologic correlation. Radiographics; 2003 Nov-Dec;23(6):1613-37
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  • Medulloblastoma is the most common pediatric central nervous system malignancy and the most common primary tumor of the posterior fossa in children.
  • This highly malignant neoplasm occurs more frequently in males and usually before 10 years of age.
  • Clinical symptoms and signs are generally brief, typically less than 3 months in duration, and reflect the strong predilection of this tumor to arise within the cerebellum, most often in the vermis.
  • Surgical resection, radiation therapy, and chemotherapy have substantially lowered the mortality associated with this tumor, with 5-year survival rates now commonly well above 50%.
  • Still, both dissemination at the time of diagnosis and recurrence remain obstacles in achieving a cure.
  • The tumor has characteristic hyperattenuation on unenhanced computed tomographic scans that reflects the high nuclear-cytoplasmic ratio seen at histologic analysis.
  • The tumor typically appears heterogeneous on images, findings that are related to cyst formation, hemorrhage, and calcification and that are even more pronounced with magnetic resonance (MR) imaging.
  • Evidence of leptomeningeal metastatic spread is present in 33% of all cases at the time of diagnosis and is well evaluated with contrast-enhanced MR imaging of the brain and the spine.
  • With continued research, treatment of these common neoplasms should improve, perhaps even achieving a cure in the future.
  • [MeSH-major] Cerebellar Neoplasms / radiography. Magnetic Resonance Imaging. Medulloblastoma / radiography. Tomography, X-Ray Computed
  • [MeSH-minor] Adolescent. Adult. Aged. Child. Child, Preschool. Female. Humans. Infant. Male. Meninges / pathology. Middle Aged. Neoplasm Invasiveness. Prognosis. Survival Rate

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  • (PMID = 14615567.001).
  • [ISSN] 1527-1323
  • [Journal-full-title] Radiographics : a review publication of the Radiological Society of North America, Inc
  • [ISO-abbreviation] Radiographics
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 102
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21. Mulne AF, Ducore JM, Elterman RD, Friedman HS, Krischer JP, Kun LE, Shuster JJ, Kadota RP: Oral methotrexate for recurrent brain tumors in children: a Pediatric Oncology Group study. J Pediatr Hematol Oncol; 2000 Jan-Feb;22(1):41-4
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  • [Title] Oral methotrexate for recurrent brain tumors in children: a Pediatric Oncology Group study.
  • PURPOSE: Children with recurrent or progressive central nervous system (CNS) tumors have an unfavorable prognosis.
  • Based on Pediatric Oncology Group (POG) institutional pilot data, low-dose oral methotrexate (MTX) was studied.
  • Patients in six different brain tumor strata were accrued.
  • RESULTS: The response rates (complete or partial responses) were as follows: astrocytoma 2 of 10, malignant glioma 1 of 19, medulloblastoma 0 of 18, brainstem tumor 0 of 12, ependymoma 1 of 7, and miscellaneous histologic types 0 of 12.
  • CONCLUSION: Low-dose oral MTX showed no significant activity against malignant glioma, medulloblastoma, brainstem tumors, and miscellaneous histologic types.
  • This regimen will not be recommended for front-line therapy.
  • [MeSH-major] Antimetabolites, Antineoplastic / therapeutic use. Brain Neoplasms / drug therapy. Methotrexate / therapeutic use. Neoplasm Recurrence, Local / drug therapy

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  • (PMID = 10695820.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA-03161; United States / NCI NIH HHS / CA / CA-29691; United States / NCI NIH HHS / CA / CA-69177; etc
  • [Publication-type] Clinical Trial; Clinical Trial, Phase II; Journal Article; Multicenter Study; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] UNITED STATES
  • [Chemical-registry-number] 0 / Antimetabolites, Antineoplastic; YL5FZ2Y5U1 / Methotrexate
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22. Sasani M, Oktenoglu T, Ozer AF, Sarioglu AC: Giant supratentorial atypical teratoid/rhabdoid tumor presentation: a case of a five-year-old child with favorable outcome and review of the literature. Pediatr Neurosurg; 2007;43(2):149-54
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  • [Title] Giant supratentorial atypical teratoid/rhabdoid tumor presentation: a case of a five-year-old child with favorable outcome and review of the literature.
  • Atypical teratoid/rhabdoid tumor of the central nervous system is a highly malignant neoplasm and that usually arises in the posterior fossa, survival from this is frequently poor.
  • We present a unique case in a 21-month-old girl who had an atypical teratoid/rhabdoid tumor with cystic components located in the right fronto-parietal lobe.
  • The patient underwent radical surgical intervention followed by chemotherapy.
  • Two years later at the last follow-up visit, there was no evidence of a tumor relapse on MRI, and the examination was symptom free.
  • It is possible the favorable outcome of the patient resulted from a rapid diagnosis, prompt management, radical surgical intervention and aggressive chemotherapy.
  • [MeSH-major] Frontal Lobe / surgery. Parietal Lobe / surgery. Rhabdoid Tumor / surgery. Supratentorial Neoplasms / surgery. Teratoma / surgery
  • [MeSH-minor] Actins / analysis. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Biomarkers, Tumor / analysis. Chemotherapy, Adjuvant. Combined Modality Therapy. Diagnosis, Differential. Female. Follow-Up Studies. Glial Fibrillary Acidic Protein / analysis. Humans. Infant. Keratins / analysis. Magnetic Resonance Imaging. Microsurgery. Mitotic Index. Necrosis. Neurologic Examination. Vimentin / analysis

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  • [Copyright] Copyright (c) 2007 S. Karger AG, Basel.
  • (PMID = 17337931.001).
  • [ISSN] 1016-2291
  • [Journal-full-title] Pediatric neurosurgery
  • [ISO-abbreviation] Pediatr Neurosurg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Actins; 0 / Biomarkers, Tumor; 0 / Glial Fibrillary Acidic Protein; 0 / Vimentin; 68238-35-7 / Keratins
  • [Number-of-references] 14
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23. Koksal Y, Toy H, Unal E, Baysal T, Esen H, Paksoy Y, Ustun ME: Pilocytic astrocytoma developing at the site of a previously treated medulloblastoma in a child. Childs Nerv Syst; 2008 Mar;24(3):289-92
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  • BACKGROUND: After the achieved high cure rates, the survivors of medulloblastoma have come to face other complications associated with treatment regimes.
  • One of these complications is secondary malignant neoplasm (SMN), which is rare but generally fatal.
  • CASE HISTORY: We report a case of an 8-year-old girl in whom a pilocytic astrocytoma developed at the site of previously excised medulloblastoma 26 months earlier.
  • The patient had then received postoperative radiotherapy and chemotherapy for the treatment of medulloblastoma.
  • Twenty-five months after the cessation of treatment, she had no complaint and physical examination was unremarkable, but a mass in the operation region was detected.
  • Rigorous and prolonged follow-up of patients with central nervous system (CNS) tumors is warrant.
  • [MeSH-major] Astrocytoma / pathology. Brain Neoplasms / pathology. Medulloblastoma / surgery. Neoplasms, Second Primary / pathology
  • [MeSH-minor] Child. Female. Humans. Treatment Outcome

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  • (PMID = 17657496.001).
  • [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; Journal Article
  • [Publication-country] Germany
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24. Spiller SE, Ravanpay AC, Hahn AW, Olson JM: Suberoylanilide hydroxamic acid is effective in preclinical studies of medulloblastoma. J Neurooncol; 2006 Sep;79(3):259-70
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  • However, little information has been reported on the effects of SAHA on central nervous system (CNS) tumors including medulloblastoma, the most common malignant brain tumor in children.
  • EXPERIMENTAL DESIGN AND RESULTS: Tissue culture studies were performed treating primary human fibroblasts, established medulloblastoma cell lines, and primary human medulloblastoma tumors with SAHA.
  • In athymic mice with medulloblastoma xenograft tumors, oral SAHA resulted in apoptosis of tumor tissue and significantly slowed tumor growth.
  • In the ND2:Smo transgenic mouse medulloblastoma model, SAHA treatment caused significant apoptosis in these cerebellar tumors.
  • CONCLUSIONS: SAHA effectively induces cell death in established medulloblastoma cell lines, human patient primary tumor cultures, medulloblastoma xenografts and intracranial spontaneous medulloblastomas.
  • These findings support the advancement of SAHA to pediatric clinical trials.
  • [MeSH-major] Antineoplastic Agents / pharmacology. Cerebellar Neoplasms / drug therapy. Hydroxamic Acids / pharmacology. Medulloblastoma / drug therapy
  • [MeSH-minor] Animals. Apoptosis / drug effects. Cells, Cultured. Child. Fibroblasts / drug effects. Humans. In Situ Nick-End Labeling. Mice. Mice, Nude. Neoplasm Transplantation. Neoplasms, Experimental / drug therapy

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  • (PMID = 16645722.001).
  • [ISSN] 0167-594X
  • [Journal-full-title] Journal of neuro-oncology
  • [ISO-abbreviation] J. Neurooncol.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA112350-01
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Hydroxamic Acids; 58IFB293JI / vorinostat
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25. Haw C, Steinbok P: Ventriculoscope tract recurrence after endoscopic biopsy of pineal germinoma. Pediatr Neurosurg; 2001 Apr;34(4):215-7
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  • Recurrence along an endoscope tract has been described after endoscopic biopsy and resection of malignant tumors arising in multiple organ systems.
  • We describe a case of tract recurrence following the ventriculoscopic biopsy of a central nervous system tumor.
  • Serum and CSF markers were negative at initial presentation and at the time of recurrence.
  • The potential for tract recurrence and CSF dissemination should be considered following the endoscopic biopsy of pineal germ cell tumors as this could affect the extent of radiotherapy and/or chemotherapy.
  • [MeSH-minor] Adolescent. Biopsy. Humans. Magnetic Resonance Imaging. Male. Neoplasm Recurrence, Local. Pineal Gland / diagnostic imaging. Pineal Gland / pathology. Third Ventricle / surgery. Tomography, X-Ray Computed

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  • [Copyright] Copyright 2001 S. Karger AG, Basel
  • (PMID = 11359115.001).
  • [ISSN] 1016-2291
  • [Journal-full-title] Pediatric neurosurgery
  • [ISO-abbreviation] Pediatr Neurosurg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Switzerland
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26. Gow KW, Lensing S, Hill DA, Krasin MJ, McCarville MB, Rai SN, Zacher M, Spunt SL, Strickland DK, Hudson MM: Thyroid carcinoma presenting in childhood or after treatment of childhood malignancies: An institutional experience and review of the literature. J Pediatr Surg; 2003 Nov;38(11):1574-80
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Thyroid carcinoma presenting in childhood or after treatment of childhood malignancies: An institutional experience and review of the literature.
  • METHODS: The authors reviewed the medical records of 8 children with PTM and 17 children with STM referred to St Jude Children's Research Hospital between February 1962 and February 2002 for evaluation and treatment of malignant thyroid carcinoma.
  • Seventeen patients had thyroid carcinoma as a second malignant neoplasm after treatment for acute lymphoblastic leukemia (n = 6), Hodgkin's disease (n = 5), central nervous system tumor (n = 2), Wilms' tumor (n = 1), retinoblastoma (n = 1), non-Hodgkin's lymphoma (n = 1), or neuroblastoma (n = 1).
  • Twelve of the 17 patients (70.6%) had received radiation to the thyroid gland during therapy for the primary cancer.
  • At the time of this report, all 17 patients are alive and in continue to be free of disease.
  • CONCLUSIONS: Pediatric thyroid carcinoma is uncommon and responds well to current therapy.
  • Given the limited period of follow-up of our cohort of secondary malignant thyroid tumors that arise after childhood cancer, these lesions appear to have similar presentations and outcomes when compared with primary carcinomas and can therefore be managed in the same manner.
  • [MeSH-major] Adenocarcinoma, Follicular / epidemiology. Carcinoma, Papillary / epidemiology. Neoplasms, Second Primary / epidemiology. Thyroid Neoplasms / epidemiology
  • [MeSH-minor] Adolescent. Adult. Child. Cohort Studies. Combined Modality Therapy. Female. Humans. Iodine Radioisotopes / therapeutic use. Lung Neoplasms / secondary. Lymphatic Metastasis. Male. Neoplasms / drug therapy. Neoplasms / radiotherapy. Neoplasms, Radiation-Induced / epidemiology. Retrospective Studies. Tennessee / epidemiology. Thyroidectomy. Treatment Outcome

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  • (PMID = 14614703.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA 21765
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Iodine Radioisotopes
  • [Number-of-references] 49
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