Treatment of young children with CNS-primitive neuroectodermal tumors/pineoblastomas in the prospective multicenter trial HIT 2000 using different chemotherapy regimens and radiotherapy.
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Treatment of young children with CNS-primitive neuroectodermal tumors/pineoblastomas in the prospective multicenter trial HIT 2000 using different chemotherapy regimens and radiotherapy. / Friedrich, Carsten; von Bueren, André O; von Hoff, Katja; Gerber, Nicolas U; Ottensmeier, Holger; Deinlein, Frank; Benesch, Martin; Kwiecien, Robert; Pietsch, Torsten; Warmuth-Metz, Monika; Faldum, Andreas; Kuehl, Joachim; Kortmann, Rolf D; Rutkowski, Stefan.
in: NEURO-ONCOLOGY, Jahrgang 15, Nr. 2, 2, 2013, S. 224-234.Publikationen: SCORING: Beitrag in Fachzeitschrift/Zeitung › SCORING: Zeitschriftenaufsatz › Forschung › Begutachtung
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TY - JOUR
T1 - Treatment of young children with CNS-primitive neuroectodermal tumors/pineoblastomas in the prospective multicenter trial HIT 2000 using different chemotherapy regimens and radiotherapy.
AU - Friedrich, Carsten
AU - von Bueren, André O
AU - von Hoff, Katja
AU - Gerber, Nicolas U
AU - Ottensmeier, Holger
AU - Deinlein, Frank
AU - Benesch, Martin
AU - Kwiecien, Robert
AU - Pietsch, Torsten
AU - Warmuth-Metz, Monika
AU - Faldum, Andreas
AU - Kuehl, Joachim
AU - Kortmann, Rolf D
AU - Rutkowski, Stefan
PY - 2013
Y1 - 2013
N2 - BACKGROUND: Especially in young children, primitive neuroectodermal tumors of the central nervous system (CNS-PNET) and pineoblastomas are associated with an unfavorable outcome, and only a few prospective trials have been conducted thus far.METHODS: From January 2001 through January 2005, 17 eligible children aged <4 years with CNS-PNET not otherwise specified (n = 8), ependymoblastoma (n = 1), or pineoblastoma (n = 8) confirmed by central review were prospectively treated in the trial HIT 2000. In nonmetastatic disease (n = 11), up to 5 postoperative cycles of HIT-SKK systemic multiagent chemotherapy (8 months duration), followed by craniospinal radiotherapy (CSI), were given. In metastatic disease (M1-M3, n = 6), treatment consisted of a shorter induction chemotherapy (2-3 months) with carboplatin and etoposide, followed by tandem high-dose chemotherapy (HDCT) in case of good response to induction. During induction and HDCT, patients received intraventricular methotrexate. CSI was applied to all patients with poor response to induction or residual disease after HDCT and was optional for patients with residual disease before HDCT.RESULTS: Five-year event-free survival and overall survival rates ± standard error for all eligible patients were 24% ± 10% and 40% ± 12%, respectively (median follow-up of survivors: 8.3 years). Only one patient with nonmetastatic disease remained free of relapse/progressive disease during induction. Three of 6 patients with metastatic disease responded to induction and received tandem-HDCT, followed by preventive CSI, and remain in continuous complete remission.CONCLUSIONS: Short intensive induction chemotherapy followed by tandem-HDCT in young children with CNS-PNET/pineoblastomas seems to be superior to the prolonged and less intensive induction regimen.
AB - BACKGROUND: Especially in young children, primitive neuroectodermal tumors of the central nervous system (CNS-PNET) and pineoblastomas are associated with an unfavorable outcome, and only a few prospective trials have been conducted thus far.METHODS: From January 2001 through January 2005, 17 eligible children aged <4 years with CNS-PNET not otherwise specified (n = 8), ependymoblastoma (n = 1), or pineoblastoma (n = 8) confirmed by central review were prospectively treated in the trial HIT 2000. In nonmetastatic disease (n = 11), up to 5 postoperative cycles of HIT-SKK systemic multiagent chemotherapy (8 months duration), followed by craniospinal radiotherapy (CSI), were given. In metastatic disease (M1-M3, n = 6), treatment consisted of a shorter induction chemotherapy (2-3 months) with carboplatin and etoposide, followed by tandem high-dose chemotherapy (HDCT) in case of good response to induction. During induction and HDCT, patients received intraventricular methotrexate. CSI was applied to all patients with poor response to induction or residual disease after HDCT and was optional for patients with residual disease before HDCT.RESULTS: Five-year event-free survival and overall survival rates ± standard error for all eligible patients were 24% ± 10% and 40% ± 12%, respectively (median follow-up of survivors: 8.3 years). Only one patient with nonmetastatic disease remained free of relapse/progressive disease during induction. Three of 6 patients with metastatic disease responded to induction and received tandem-HDCT, followed by preventive CSI, and remain in continuous complete remission.CONCLUSIONS: Short intensive induction chemotherapy followed by tandem-HDCT in young children with CNS-PNET/pineoblastomas seems to be superior to the prolonged and less intensive induction regimen.
KW - Humans
KW - Male
KW - Female
KW - Quality of Life
KW - Neuropsychological Tests
KW - Prospective Studies
KW - Prognosis
KW - Survival Rate
KW - Follow-Up Studies
KW - Child, Preschool
KW - Infant
KW - Infant, Newborn
KW - Chemoradiotherapy
KW - Antineoplastic Combined Chemotherapy Protocols/therapeutic use
KW - Carboplatin/administration & dosage
KW - Cyclophosphamide/administration & dosage
KW - Etoposide/administration & dosage
KW - Methotrexate/administration & dosage
KW - Vincristine/administration & dosage
KW - Neoplasm Grading
KW - Brain Neoplasms/mortality/pathology/therapy
KW - Cranial Irradiation
KW - Neoplasm, Residual/mortality/pathology/therapy
KW - Neuroectodermal Tumors, Primitive/mortality/pathology/therapy
KW - Pineal Gland/drug effects/pathology/radiation effects
KW - Pinealoma/mortality/pathology/therapy
KW - Humans
KW - Male
KW - Female
KW - Quality of Life
KW - Neuropsychological Tests
KW - Prospective Studies
KW - Prognosis
KW - Survival Rate
KW - Follow-Up Studies
KW - Child, Preschool
KW - Infant
KW - Infant, Newborn
KW - Chemoradiotherapy
KW - Antineoplastic Combined Chemotherapy Protocols/therapeutic use
KW - Carboplatin/administration & dosage
KW - Cyclophosphamide/administration & dosage
KW - Etoposide/administration & dosage
KW - Methotrexate/administration & dosage
KW - Vincristine/administration & dosage
KW - Neoplasm Grading
KW - Brain Neoplasms/mortality/pathology/therapy
KW - Cranial Irradiation
KW - Neoplasm, Residual/mortality/pathology/therapy
KW - Neuroectodermal Tumors, Primitive/mortality/pathology/therapy
KW - Pineal Gland/drug effects/pathology/radiation effects
KW - Pinealoma/mortality/pathology/therapy
U2 - 10.1093/neuonc/nos292
DO - 10.1093/neuonc/nos292
M3 - SCORING: Journal article
C2 - 23223339
VL - 15
SP - 224
EP - 234
JO - NEURO-ONCOLOGY
JF - NEURO-ONCOLOGY
SN - 1522-8517
IS - 2
M1 - 2
ER -
