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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.
Cancer. 2009 Jul 01; 115(13):2956-63.C

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-PNET who were referred for myeloablative chemotherapy and autologous hematopoietic progenitor cell rescue at Childrens Hospital Los Angeles.

RESULTS

Thirty-three children were referred for myeloablative chemotherapy: Fourteen of those children were never transplanted because of pre-transplant adverse events, and 19, including 6 without and 13 with previous irradiation, underwent transplant. 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 radiotherapy were able to cure most children who had radiotherapy-naive, chemoresponsive recurrences. Children who developed recurrences after craniospinal radiotherapy had poorer outcomes; however, cure was possible in those who had good prognostic features at presentation, chemoresponsive recurrences, and a long interval between initial radiotherapy and myeloablative chemotherapy.

Authors+Show Affiliations

Division of Hematology/Oncology, Childrens Hospital Los Angeles and Keck School of Medicine, University of Southern California, Los Angeles, California, USA. abutturini@chla.usc.eduNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article

Language

eng

PubMed ID

19402050

Citation

Butturini, Anna M., et al. "High-dose Chemotherapy and Autologous Hematopoietic Progenitor Cell Rescue in Children With Recurrent Medulloblastoma and Supratentorial Primitive Neuroectodermal Tumors: the Impact of Prior Radiotherapy On Outcome." Cancer, vol. 115, no. 13, 2009, pp. 2956-63.
Butturini AM, Jacob M, Aguajo J, et al. High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors: the impact of prior radiotherapy on outcome. Cancer. 2009;115(13):2956-63.
Butturini, A. M., Jacob, M., Aguajo, J., Vander-Walde, N. A., Villablanca, J., Jubran, R., Erdreich-Epstein, A., Marachelian, A., Dhall, G., & Finlay, J. L. (2009). High-dose chemotherapy and autologous hematopoietic progenitor cell rescue in children with recurrent medulloblastoma and supratentorial primitive neuroectodermal tumors: the impact of prior radiotherapy on outcome. Cancer, 115(13), 2956-63. https://doi.org/10.1002/cncr.24341
Butturini AM, et al. High-dose Chemotherapy and Autologous Hematopoietic Progenitor Cell Rescue in Children With Recurrent Medulloblastoma and Supratentorial Primitive Neuroectodermal Tumors: the Impact of Prior Radiotherapy On Outcome. Cancer. 2009 Jul 1;115(13):2956-63. PubMed PMID: 19402050.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - 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. AU - Butturini,Anna M, AU - Jacob,Mary, AU - Aguajo,Jennifer, AU - Vander-Walde,Noam A, AU - Villablanca,Judy, AU - Jubran,Rima, AU - Erdreich-Epstein,Anat, AU - Marachelian,Araz, AU - Dhall,Girish, AU - Finlay,Jonathan L, PY - 2009/4/30/entrez PY - 2009/4/30/pubmed PY - 2009/7/23/medline SP - 2956 EP - 63 JF - Cancer JO - Cancer VL - 115 IS - 13 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-PNET who were referred for myeloablative chemotherapy and autologous hematopoietic progenitor cell rescue at Childrens Hospital Los Angeles. RESULTS: Thirty-three children were referred for myeloablative chemotherapy: Fourteen of those children were never transplanted because of pre-transplant adverse events, and 19, including 6 without and 13 with previous irradiation, underwent transplant. 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 radiotherapy were able to cure most children who had radiotherapy-naive, chemoresponsive recurrences. Children who developed recurrences after craniospinal radiotherapy had poorer outcomes; however, cure was possible in those who had good prognostic features at presentation, chemoresponsive recurrences, and a long interval between initial radiotherapy and myeloablative chemotherapy. SN - 0008-543X UR - https://www.unboundmedicine.com/medline/citation/19402050/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_ L2 - https://doi.org/10.1002/cncr.24341 DB - PRIME DP - Unbound Medicine ER -