High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors

The impact of prior radiotherapy on outcome

Anna M. Butturini, Mary Jacob, Jennifer Aguajo, Noam Vanderwalde, Judy Villablanca, Rima Jubran, Anat Erdreich-Epstein, Araz Marachelian, Girish Dhall, Jonathan L. Finlay

Research output: Contribution to journalArticle

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Abstract

BACKGROUND: The role of myeloablative chemotherapy in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors (MB/ST-PNET) is controversial, in particular in patients who develop recurrent disease after craniospinal radiotherapy. METHODS: In this retrospective analysis, the authors investigated the outcome of children with recurrent MB/ST-PNETwhowere referred for myeloablative chemotherapy and autologous hematopoietic progenitor cell rescue at Childrens Hospital Los Angeles. RESULTS: Thirty-three children were referred for myeloablative chemotherapy: Fourteen of those children were never transplanted because of pre-transplant adverse events, and 19, including 6 without and 13 with previous irradiation, underwent transplant. Conditioning regimens included a backbone of thiotepa, which was given either in a single cycle or in multiple sequential cycles. The 3-year post-transplant event-free survival rate in unirradiated versus previously irradiated children was 83%±15% versus 20%±12%, respectively (P=.04). One child who had never been exposed to radiotherapy died of toxicity; the other children received post-transplant radiotherapy and remained disease free. Nine previously irradiated children experienced 4 toxic deaths and 6 tumor recurrences (1 patient had both): An interval of <1 year between initial radiotherapy and myeloablative chemotherapy predicted a greater risk of toxic death (P=.02), whereas a history of meningeal metastases at diagnosis and a poor response to the initial rescue therapy predicted a greater risk of post-transplant recurrence (P=.03 and P=.08, respectively). CONCLUSIONS: Myeloablative doses of thiotepa-based chemotherapy and radiotherapywere able to curemost childrenwho had radiotherapynaive, chemoresponsive recurrences. Children who developed recurrences after craniospinal radiotherapy had poorer outcomes; however, cure was possible in those who had good prognostic features at presentation, chemoresponsive recurrences, and a long interval between initial radiotherapy andmyeloablative chemotherapy.

Original languageEnglish (US)
Pages (from-to)2956-2963
Number of pages8
JournalCancer
Volume115
Issue number13
DOIs
StatePublished - Jul 1 2009

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Primitive Neuroectodermal Tumors
Medulloblastoma
Hematopoietic Stem Cells
Radiotherapy
Drug Therapy
Transplants
Recurrence
Thiotepa
Poisons
Los Angeles
Disease-Free Survival
Survival Rate
Neoplasm Metastasis

All Science Journal Classification (ASJC) codes

  • Oncology
  • Cancer Research

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High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors : The impact of prior radiotherapy on outcome. / Butturini, Anna M.; Jacob, Mary; Aguajo, Jennifer; Vanderwalde, Noam; Villablanca, Judy; Jubran, Rima; Erdreich-Epstein, Anat; Marachelian, Araz; Dhall, Girish; Finlay, Jonathan L.

In: Cancer, Vol. 115, No. 13, 01.07.2009, p. 2956-2963.

Research output: Contribution to journalArticle

Butturini, Anna M. ; Jacob, Mary ; Aguajo, Jennifer ; Vanderwalde, Noam ; Villablanca, Judy ; Jubran, Rima ; Erdreich-Epstein, Anat ; Marachelian, Araz ; Dhall, Girish ; Finlay, Jonathan L. / High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors : The impact of prior radiotherapy on outcome. In: Cancer. 2009 ; Vol. 115, No. 13. pp. 2956-2963.
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abstract = "BACKGROUND: The role of myeloablative chemotherapy in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors (MB/ST-PNET) is controversial, in particular in patients who develop recurrent disease after craniospinal radiotherapy. METHODS: In this retrospective analysis, the authors investigated the outcome of children with recurrent MB/ST-PNETwhowere referred for myeloablative chemotherapy and autologous hematopoietic progenitor cell rescue at Childrens Hospital Los Angeles. RESULTS: Thirty-three children were referred for myeloablative chemotherapy: Fourteen of those children were never transplanted because of pre-transplant adverse events, and 19, including 6 without and 13 with previous irradiation, underwent transplant. Conditioning regimens included a backbone of thiotepa, which was given either in a single cycle or in multiple sequential cycles. The 3-year post-transplant event-free survival rate in unirradiated versus previously irradiated children was 83{\%}±15{\%} versus 20{\%}±12{\%}, respectively (P=.04). One child who had never been exposed to radiotherapy died of toxicity; the other children received post-transplant radiotherapy and remained disease free. Nine previously irradiated children experienced 4 toxic deaths and 6 tumor recurrences (1 patient had both): An interval of <1 year between initial radiotherapy and myeloablative chemotherapy predicted a greater risk of toxic death (P=.02), whereas a history of meningeal metastases at diagnosis and a poor response to the initial rescue therapy predicted a greater risk of post-transplant recurrence (P=.03 and P=.08, respectively). CONCLUSIONS: Myeloablative doses of thiotepa-based chemotherapy and radiotherapywere able to curemost childrenwho had radiotherapynaive, chemoresponsive recurrences. Children who developed recurrences after craniospinal radiotherapy had poorer outcomes; however, cure was possible in those who had good prognostic features at presentation, chemoresponsive recurrences, and a long interval between initial radiotherapy andmyeloablative chemotherapy.",
author = "Butturini, {Anna M.} and Mary Jacob and Jennifer Aguajo and Noam Vanderwalde and Judy Villablanca and Rima Jubran and Anat Erdreich-Epstein and Araz Marachelian and Girish Dhall and Finlay, {Jonathan L.}",
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T1 - High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors

T2 - The impact of prior radiotherapy on outcome

AU - Butturini, Anna M.

AU - Jacob, Mary

AU - Aguajo, Jennifer

AU - Vanderwalde, Noam

AU - Villablanca, Judy

AU - Jubran, Rima

AU - Erdreich-Epstein, Anat

AU - Marachelian, Araz

AU - Dhall, Girish

AU - Finlay, Jonathan L.

PY - 2009/7/1

Y1 - 2009/7/1

N2 - BACKGROUND: The role of myeloablative chemotherapy in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors (MB/ST-PNET) is controversial, in particular in patients who develop recurrent disease after craniospinal radiotherapy. METHODS: In this retrospective analysis, the authors investigated the outcome of children with recurrent MB/ST-PNETwhowere referred for myeloablative chemotherapy and autologous hematopoietic progenitor cell rescue at Childrens Hospital Los Angeles. RESULTS: Thirty-three children were referred for myeloablative chemotherapy: Fourteen of those children were never transplanted because of pre-transplant adverse events, and 19, including 6 without and 13 with previous irradiation, underwent transplant. Conditioning regimens included a backbone of thiotepa, which was given either in a single cycle or in multiple sequential cycles. The 3-year post-transplant event-free survival rate in unirradiated versus previously irradiated children was 83%±15% versus 20%±12%, respectively (P=.04). One child who had never been exposed to radiotherapy died of toxicity; the other children received post-transplant radiotherapy and remained disease free. Nine previously irradiated children experienced 4 toxic deaths and 6 tumor recurrences (1 patient had both): An interval of <1 year between initial radiotherapy and myeloablative chemotherapy predicted a greater risk of toxic death (P=.02), whereas a history of meningeal metastases at diagnosis and a poor response to the initial rescue therapy predicted a greater risk of post-transplant recurrence (P=.03 and P=.08, respectively). CONCLUSIONS: Myeloablative doses of thiotepa-based chemotherapy and radiotherapywere able to curemost childrenwho had radiotherapynaive, chemoresponsive recurrences. Children who developed recurrences after craniospinal radiotherapy had poorer outcomes; however, cure was possible in those who had good prognostic features at presentation, chemoresponsive recurrences, and a long interval between initial radiotherapy andmyeloablative chemotherapy.

AB - BACKGROUND: The role of myeloablative chemotherapy in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors (MB/ST-PNET) is controversial, in particular in patients who develop recurrent disease after craniospinal radiotherapy. METHODS: In this retrospective analysis, the authors investigated the outcome of children with recurrent MB/ST-PNETwhowere referred for myeloablative chemotherapy and autologous hematopoietic progenitor cell rescue at Childrens Hospital Los Angeles. RESULTS: Thirty-three children were referred for myeloablative chemotherapy: Fourteen of those children were never transplanted because of pre-transplant adverse events, and 19, including 6 without and 13 with previous irradiation, underwent transplant. Conditioning regimens included a backbone of thiotepa, which was given either in a single cycle or in multiple sequential cycles. The 3-year post-transplant event-free survival rate in unirradiated versus previously irradiated children was 83%±15% versus 20%±12%, respectively (P=.04). One child who had never been exposed to radiotherapy died of toxicity; the other children received post-transplant radiotherapy and remained disease free. Nine previously irradiated children experienced 4 toxic deaths and 6 tumor recurrences (1 patient had both): An interval of <1 year between initial radiotherapy and myeloablative chemotherapy predicted a greater risk of toxic death (P=.02), whereas a history of meningeal metastases at diagnosis and a poor response to the initial rescue therapy predicted a greater risk of post-transplant recurrence (P=.03 and P=.08, respectively). CONCLUSIONS: Myeloablative doses of thiotepa-based chemotherapy and radiotherapywere able to curemost childrenwho had radiotherapynaive, chemoresponsive recurrences. Children who developed recurrences after craniospinal radiotherapy had poorer outcomes; however, cure was possible in those who had good prognostic features at presentation, chemoresponsive recurrences, and a long interval between initial radiotherapy andmyeloablative chemotherapy.

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