Atypical teratoid/rhabdoid tumors in children treated with multimodal therapies

The necessity of upfront radiotherapy after surgery

Jeongshim Lee, Dong Seok Kim, Jung Woo Han, Chang-Ok Suh

Research output: Contribution to journalArticle

4 Citations (Scopus)

Abstract

Background: Atypical teratoid/rhabdoid tumor (ATRT) is a rare malignant pediatric brain tumor with a dismal prognosis. We evaluated the efficacy of multimodal therapy in children with ATRT. Procedure: Nine children diagnosed with cranial ATRT, who received multimodal therapy between 2005 and 2014, including surgical resection followed by radiotherapy (RT), systemic chemotherapy (CT), and high-dose chemotherapy/stem cell transplantation (HDCT/SCT), were analyzed retrospectively. The median age at diagnosis was 32 months. Initial treatment included surgery in eight patients (88%), CT in all (100%), RT in eight (88%), and HDCT/SCT in seven (78%). Results: The median follow-up period for survivors was 21 months. The 2-year progression-free rate was 66.7%. Two patients had progression 4 and 17 months after diagnosis. One received multimodal treatment, including surgery and upfront CT with delayed RT; the other underwent surgery and upfront CT without RT. The 2-year event-free survival and overall survival rates were 46.7% and 62.2%, respectively. Hematologic toxicity of grade 3 or more was observed in six patients treated with HDCT/SCT and two who underwent craniospinal irradiation. Deaths were attributed to progressive disease (n = 2) and treatment-related toxicity (n = 2) from sepsis and acute respiratory failure after CT and HDCT/SCT. Conclusions: Maximal safe resection in conjunction with upfront RT is a reasonable multimodal treatment in patients with ATRT for prolonging progression-free survival. Further research may help determine the optimal parameters for reducing treatment toxicity, such as intensity of HDCT/SCT and the RT field.

Original languageEnglish
Article numbere26663
JournalPediatric Blood and Cancer
Volume64
Issue number12
DOIs
Publication statusPublished - 2017 Dec 1

Fingerprint

Radiotherapy
Drug Therapy
Stem Cell Transplantation
Therapeutics
Combined Modality Therapy
Disease-Free Survival
Craniospinal Irradiation
Atypical Teratoid Tumor
Typical Teratoid Rhabdoid Tumor
Brain Neoplasms
Respiratory Insufficiency
Survivors
Sepsis
Survival Rate
Pediatrics
Research

All Science Journal Classification (ASJC) codes

  • Pediatrics, Perinatology, and Child Health
  • Hematology
  • Oncology

Cite this

@article{ea434c9c106b4c20867e148a9e8ada68,
title = "Atypical teratoid/rhabdoid tumors in children treated with multimodal therapies: The necessity of upfront radiotherapy after surgery",
abstract = "Background: Atypical teratoid/rhabdoid tumor (ATRT) is a rare malignant pediatric brain tumor with a dismal prognosis. We evaluated the efficacy of multimodal therapy in children with ATRT. Procedure: Nine children diagnosed with cranial ATRT, who received multimodal therapy between 2005 and 2014, including surgical resection followed by radiotherapy (RT), systemic chemotherapy (CT), and high-dose chemotherapy/stem cell transplantation (HDCT/SCT), were analyzed retrospectively. The median age at diagnosis was 32 months. Initial treatment included surgery in eight patients (88{\%}), CT in all (100{\%}), RT in eight (88{\%}), and HDCT/SCT in seven (78{\%}). Results: The median follow-up period for survivors was 21 months. The 2-year progression-free rate was 66.7{\%}. Two patients had progression 4 and 17 months after diagnosis. One received multimodal treatment, including surgery and upfront CT with delayed RT; the other underwent surgery and upfront CT without RT. The 2-year event-free survival and overall survival rates were 46.7{\%} and 62.2{\%}, respectively. Hematologic toxicity of grade 3 or more was observed in six patients treated with HDCT/SCT and two who underwent craniospinal irradiation. Deaths were attributed to progressive disease (n = 2) and treatment-related toxicity (n = 2) from sepsis and acute respiratory failure after CT and HDCT/SCT. Conclusions: Maximal safe resection in conjunction with upfront RT is a reasonable multimodal treatment in patients with ATRT for prolonging progression-free survival. Further research may help determine the optimal parameters for reducing treatment toxicity, such as intensity of HDCT/SCT and the RT field.",
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Atypical teratoid/rhabdoid tumors in children treated with multimodal therapies : The necessity of upfront radiotherapy after surgery. / Lee, Jeongshim; Kim, Dong Seok; Han, Jung Woo; Suh, Chang-Ok.

In: Pediatric Blood and Cancer, Vol. 64, No. 12, e26663, 01.12.2017.

Research output: Contribution to journalArticle

TY - JOUR

T1 - Atypical teratoid/rhabdoid tumors in children treated with multimodal therapies

T2 - The necessity of upfront radiotherapy after surgery

AU - Lee, Jeongshim

AU - Kim, Dong Seok

AU - Han, Jung Woo

AU - Suh, Chang-Ok

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N2 - Background: Atypical teratoid/rhabdoid tumor (ATRT) is a rare malignant pediatric brain tumor with a dismal prognosis. We evaluated the efficacy of multimodal therapy in children with ATRT. Procedure: Nine children diagnosed with cranial ATRT, who received multimodal therapy between 2005 and 2014, including surgical resection followed by radiotherapy (RT), systemic chemotherapy (CT), and high-dose chemotherapy/stem cell transplantation (HDCT/SCT), were analyzed retrospectively. The median age at diagnosis was 32 months. Initial treatment included surgery in eight patients (88%), CT in all (100%), RT in eight (88%), and HDCT/SCT in seven (78%). Results: The median follow-up period for survivors was 21 months. The 2-year progression-free rate was 66.7%. Two patients had progression 4 and 17 months after diagnosis. One received multimodal treatment, including surgery and upfront CT with delayed RT; the other underwent surgery and upfront CT without RT. The 2-year event-free survival and overall survival rates were 46.7% and 62.2%, respectively. Hematologic toxicity of grade 3 or more was observed in six patients treated with HDCT/SCT and two who underwent craniospinal irradiation. Deaths were attributed to progressive disease (n = 2) and treatment-related toxicity (n = 2) from sepsis and acute respiratory failure after CT and HDCT/SCT. Conclusions: Maximal safe resection in conjunction with upfront RT is a reasonable multimodal treatment in patients with ATRT for prolonging progression-free survival. Further research may help determine the optimal parameters for reducing treatment toxicity, such as intensity of HDCT/SCT and the RT field.

AB - Background: Atypical teratoid/rhabdoid tumor (ATRT) is a rare malignant pediatric brain tumor with a dismal prognosis. We evaluated the efficacy of multimodal therapy in children with ATRT. Procedure: Nine children diagnosed with cranial ATRT, who received multimodal therapy between 2005 and 2014, including surgical resection followed by radiotherapy (RT), systemic chemotherapy (CT), and high-dose chemotherapy/stem cell transplantation (HDCT/SCT), were analyzed retrospectively. The median age at diagnosis was 32 months. Initial treatment included surgery in eight patients (88%), CT in all (100%), RT in eight (88%), and HDCT/SCT in seven (78%). Results: The median follow-up period for survivors was 21 months. The 2-year progression-free rate was 66.7%. Two patients had progression 4 and 17 months after diagnosis. One received multimodal treatment, including surgery and upfront CT with delayed RT; the other underwent surgery and upfront CT without RT. The 2-year event-free survival and overall survival rates were 46.7% and 62.2%, respectively. Hematologic toxicity of grade 3 or more was observed in six patients treated with HDCT/SCT and two who underwent craniospinal irradiation. Deaths were attributed to progressive disease (n = 2) and treatment-related toxicity (n = 2) from sepsis and acute respiratory failure after CT and HDCT/SCT. Conclusions: Maximal safe resection in conjunction with upfront RT is a reasonable multimodal treatment in patients with ATRT for prolonging progression-free survival. Further research may help determine the optimal parameters for reducing treatment toxicity, such as intensity of HDCT/SCT and the RT field.

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