Atypical Teratoid/Rhabdoid Tumor of the Central Nervous System in Children under the Age of 3 Years

Purpose Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze...

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Published inCancer research and treatment Vol. 53; no. 2; pp. 378 - 388
Main Authors Park, Meerim, Han, Jung Woo, Hahn, Seung Min, Lee, Jun Ah, Kim, Joo-Young, Shin, Sang Hoon, Kim, Dong-Seok, Yoon, Hong In, Hong, Kyung Taek, Choi, Jung Yoon, Kang, Hyoung Jin, Shin, Hee Young, Phi, Ji Hoon, Kim, Seung-Ki, Lee, Ji Won, Yoo, Keon Hee, Sung, Ki Woong, Koo, Hong Hoe, Lim, Do Hoon, Shin, Hyung Jin, Kim, Hyery, Koh, Kyung-Nam, Im, Ho Joon, Ahn, Seung Do, Ra, Young-Shin, Baek, Hee-Jo, Kook, Hoon, Jung, Tae-Young, Choi, Hyoung Soo, Kim, Chae-Yong, Park, Hyeon Jin, Lyu, Chuhl Joo
Format Journal Article
LanguageEnglish
Published Korea (South) Korean Cancer Association 01.04.2021
대한암학회
Subjects
Central Nervous System Neoplasms - diagnosis
Central Nervous System Neoplasms - mortality
Central Nervous System Neoplasms - pathology
Child, Preschool
Female
Humans
Infant
Infant, Newborn
Male
Original
Progression-Free Survival
Rhabdoid Tumor - diagnosis
Rhabdoid Tumor - mortality
Rhabdoid Tumor - pathology
의학일반
ATRT
Children
Radiotherapy
High-dose chemotherapy
Online AccessGet full text
ISSN1598-2998
2005-9256
2005-9256
DOI10.4143/crt.2020.756

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Abstract Purpose Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze characteristics and clinical outcomes of ATRT in children aged less than 3 years.Materials and Methods A search of medical records from seven centers was performed between January 2005 and December 2016.Results Forty-three patients were enrolled. With a median follow-up of 90 months, 27 patients (64.3%) showed at least one episode of disease progression (PD). The first date of PD was at 160 days after diagnosis. The 1- and 3-year progression-free survivals (PFS) were 51.2% and 28.5%, respectively. The 1- and 3-year overall survivals were 61.9% and 38.1%, respectively. The 3-year PFS was improved from 0% in pre-2011 to 47.4% in post-2011. Excluding one patient who did not receive any further therapy after surgery, 27 patients died due to PD (n=21), treatment-related toxicity (n=5), or unknown cause (n=1). In univariate analysis, factors associated with higher 3-year PFS were no metastases, diagnosis after 2011, early adjuvant radiotherapy, and high-dose chemotherapy (HDCT). In multivariate analysis, the use of HDCT and adjuvant radiotherapy remained significant prognostic factors for PFS (both p < 0.01).Conclusion Aggressive therapy including early adjuvant radiotherapy and HDCT could be considered to improve outcomes of ATRT in children under the age of 3 years.
AbstractList Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze characteristics and clinical outcomes of ATRT in children aged less than 3 years.PURPOSEAtypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze characteristics and clinical outcomes of ATRT in children aged less than 3 years.A search of medical records from seven centers was performed between January 2005 and December 2016.MATERIALS AND METHODSA search of medical records from seven centers was performed between January 2005 and December 2016.Forty-three patients were enrolled. With a median follow-up of 90 months, 27 patients (64.3%) showed at least one episode of disease progression (PD). The first date of PD was at 160 days after diagnosis. The 1- and 3-year progression-free survivals (PFS) were 51.2% and 28.5%, respectively. The 1- and 3-year overall survivals were 61.9% and 38.1%, respectively. The 3-year PFS was improved from 0% in pre-2011 to 47.4% in post-2011. Excluding one patient who did not receive any further therapy after surgery, 27 patients died due to PD (n=21), treatment-related toxicity (n=5), or unknown cause (n=1). In univariate analysis, factors associated with higher 3-year PFS were no metastases, diagnosis after 2011, early adjuvant radiotherapy, and high-dose chemotherapy (HDCT). In multivariate analysis, the use of HDCT and adjuvant radiotherapy remained significant prognostic factors for PFS (both p < 0.01).RESULTSForty-three patients were enrolled. With a median follow-up of 90 months, 27 patients (64.3%) showed at least one episode of disease progression (PD). The first date of PD was at 160 days after diagnosis. The 1- and 3-year progression-free survivals (PFS) were 51.2% and 28.5%, respectively. The 1- and 3-year overall survivals were 61.9% and 38.1%, respectively. The 3-year PFS was improved from 0% in pre-2011 to 47.4% in post-2011. Excluding one patient who did not receive any further therapy after surgery, 27 patients died due to PD (n=21), treatment-related toxicity (n=5), or unknown cause (n=1). In univariate analysis, factors associated with higher 3-year PFS were no metastases, diagnosis after 2011, early adjuvant radiotherapy, and high-dose chemotherapy (HDCT). In multivariate analysis, the use of HDCT and adjuvant radiotherapy remained significant prognostic factors for PFS (both p < 0.01).Aggressive therapy including early adjuvant radiotherapy and HDCT could be considered to improve outcomes of ATRT in children under the age of 3 years.CONCLUSIONAggressive therapy including early adjuvant radiotherapy and HDCT could be considered to improve outcomes of ATRT in children under the age of 3 years.
Purpose Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze characteristics and clinical outcomes of ATRT in children aged less than 3 years.Materials and Methods A search of medical records from seven centers was performed between January 2005 and December 2016.Results Forty-three patients were enrolled. With a median follow-up of 90 months, 27 patients (64.3%) showed at least one episode of disease progression (PD). The first date of PD was at 160 days after diagnosis. The 1- and 3-year progression-free survivals (PFS) were 51.2% and 28.5%, respectively. The 1- and 3-year overall survivals were 61.9% and 38.1%, respectively. The 3-year PFS was improved from 0% in pre-2011 to 47.4% in post-2011. Excluding one patient who did not receive any further therapy after surgery, 27 patients died due to PD (n=21), treatment-related toxicity (n=5), or unknown cause (n=1). In univariate analysis, factors associated with higher 3-year PFS were no metastases, diagnosis after 2011, early adjuvant radiotherapy, and high-dose chemotherapy (HDCT). In multivariate analysis, the use of HDCT and adjuvant radiotherapy remained significant prognostic factors for PFS (both p < 0.01).Conclusion Aggressive therapy including early adjuvant radiotherapy and HDCT could be considered to improve outcomes of ATRT in children under the age of 3 years. KCI Citation Count: 0
Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze characteristics and clinical outcomes of ATRT in children aged less than 3 years. A search of medical records from seven centers was performed between January 2005 and December 2016. Forty-three patients were enrolled. With a median follow-up of 90 months, 27 patients (64.3%) showed at least one episode of disease progression (PD). The first date of PD was at 160 days after diagnosis. The 1- and 3-year progression-free survivals (PFS) were 51.2% and 28.5%, respectively. The 1- and 3-year overall survivals were 61.9% and 38.1%, respectively. The 3-year PFS was improved from 0% in pre-2011 to 47.4% in post-2011. Excluding one patient who did not receive any further therapy after surgery, 27 patients died due to PD (n=21), treatment-related toxicity (n=5), or unknown cause (n=1). In univariate analysis, factors associated with higher 3-year PFS were no metastases, diagnosis after 2011, early adjuvant radiotherapy, and high-dose chemotherapy (HDCT). In multivariate analysis, the use of HDCT and adjuvant radiotherapy remained significant prognostic factors for PFS (both p < 0.01). Aggressive therapy including early adjuvant radiotherapy and HDCT could be considered to improve outcomes of ATRT in children under the age of 3 years.
Purpose Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze characteristics and clinical outcomes of ATRT in children aged less than 3 years.Materials and Methods A search of medical records from seven centers was performed between January 2005 and December 2016.Results Forty-three patients were enrolled. With a median follow-up of 90 months, 27 patients (64.3%) showed at least one episode of disease progression (PD). The first date of PD was at 160 days after diagnosis. The 1- and 3-year progression-free survivals (PFS) were 51.2% and 28.5%, respectively. The 1- and 3-year overall survivals were 61.9% and 38.1%, respectively. The 3-year PFS was improved from 0% in pre-2011 to 47.4% in post-2011. Excluding one patient who did not receive any further therapy after surgery, 27 patients died due to PD (n=21), treatment-related toxicity (n=5), or unknown cause (n=1). In univariate analysis, factors associated with higher 3-year PFS were no metastases, diagnosis after 2011, early adjuvant radiotherapy, and high-dose chemotherapy (HDCT). In multivariate analysis, the use of HDCT and adjuvant radiotherapy remained significant prognostic factors for PFS (both p < 0.01).Conclusion Aggressive therapy including early adjuvant radiotherapy and HDCT could be considered to improve outcomes of ATRT in children under the age of 3 years.
Author Yoon, Hong In
Jung, Tae-Young
Lee, Jun Ah
Kim, Hyery
Lyu, Chuhl Joo
Kang, Hyoung Jin
Im, Ho Joon
Phi, Ji Hoon
Kook, Hoon
Choi, Hyoung Soo
Ahn, Seung Do
Lim, Do Hoon
Kim, Seung-Ki
Park, Meerim
Shin, Hee Young
Hahn, Seung Min
Choi, Jung Yoon
Lee, Ji Won
Koo, Hong Hoe
Kim, Chae-Yong
Kim, Joo-Young
Hong, Kyung Taek
Ra, Young-Shin
Kim, Dong-Seok
Sung, Ki Woong
Koh, Kyung-Nam
Yoo, Keon Hee
Shin, Sang Hoon
Han, Jung Woo
Shin, Hyung Jin
Baek, Hee-Jo
Park, Hyeon Jin
AuthorAffiliation 2 Department of Pediatrics, Yonsei University College of Medicine, Seoul, Korea
14 Neurosurgery, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
8 Department of Neurosurgery, Seoul National University College of Medicine, Seoul National University Cancer Research Institute, Seoul, Korea
5 Department of Neurosurgery, Yonsei University College of Medicine, Seoul, Korea
6 Radiation Oncology, Yonsei University College of Medicine, Seoul, Korea
15 Department of Pediatrics, Chonnam National University Hwasun Hospital, Chonnam National University Medical School, Hwasun, Korea
9 Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea
13 Radiation Oncology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
4 Neuro-Oncology Clinic, Center for Pediatric Cancer, National Cancer Center, Goyang, Korea
3 Department of Radiation Oncology, Center for Pediatric Cancer, National Cancer Center, Goyang, Korea
11 Ne
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Issue 2
Keywords ATRT
Children
Radiotherapy
High-dose chemotherapy
Language English
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Meerim Park and Jung Woo Han contributed equally to this work.
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Snippet Purpose Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment...
Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for...
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SubjectTerms Central Nervous System Neoplasms - diagnosis
Central Nervous System Neoplasms - mortality
Central Nervous System Neoplasms - pathology
Child, Preschool
Female
Humans
Infant
Infant, Newborn
Male
Original
Progression-Free Survival
Rhabdoid Tumor - diagnosis
Rhabdoid Tumor - mortality
Rhabdoid Tumor - pathology
의학일반
Title Atypical Teratoid/Rhabdoid Tumor of the Central Nervous System in Children under the Age of 3 Years
URI https://www.ncbi.nlm.nih.gov/pubmed/33138347
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