Second Neoplasms in Pediatric Patients with Primary Central Nervous System Tumors: The St. Jude Children's Research Hospital Experience

Alberto Broniscer, Weiming Ke, Christine E. Fuller, Jianrong Wu, Amar Gajjar, Larry E. Kun

Research output: Contribution to journalReview article

77 Citations (Scopus)

Abstract

BACKGROUND. Details on second neoplasms (SNs) following pediatric central nervous system (CNS) tumors are scant, because of the rarity of such SNs. The goal of the current study was to investigate and characterize these rare SNs. METHODS. The authors reviewed clinical and treatment data on all institutional patients age < 22 years at diagnosis of a primary CNS tumor who developed any type of SN. Patients with neurofibromatosis type 1 were excluded. Cumulative incidence rates were estimated, and putative risk factors were analyzed. RESULTS. The SNs investigated in the current study included 10 gliomas (42%), 5 meningiomas (21%), 2 desmoid tumors, 2 myelodysplastic syndromes, 2 basal cell carcinomas, 1 leukemia, 1 malignant fibrous histiocytoma, and 1 thyroid carcinoma. Twenty-one patients had previously received radiotherapy, and 12 patients had received chemotherapy. The SN was related to a genetic cause in 7 patients (29%). Eleven patients died of their SNs, including 8 patients with glioma and 2 patients with myelodysplastic syndromes. The estimated 15-year cumulative incidence rate for malignant SNs was 4%. Children with choroid plexus tumors had an estimated 10-year cumulative incidence rate of 20.2%; 2 of those patients had germline TP53 mutations. Age ≤ 2 years was a significant risk factor (P = 0.016) for development of an SN only when patients with genetic conditions were included in the analysis. No significant difference in the estimated cumulative incidence of SNs was found among patients who had received different types of therapy. CONCLUSIONS. The risk of lethal SNs after pediatric CNS tumors is small. Young patients and patients with choroid plexus tumors appear to have an increased risk of SNs that is associated with genetic factors.

Original languageEnglish (US)
Pages (from-to)2246-2252
Number of pages7
JournalCancer
Volume100
Issue number10
DOIs
StatePublished - May 15 2004

Fingerprint

Central Nervous System Neoplasms
Second Primary Neoplasms
Pediatrics
Research
Choroid Plexus Neoplasms
Myelodysplastic Syndromes
Incidence
Glioma
Aggressive Fibromatosis
Malignant Fibrous Histiocytoma
Neurofibromatosis 1
Germ-Line Mutation
Basal Cell Carcinoma
Meningioma
Thyroid Neoplasms

Keywords

  • Brain
  • Central nervous system
  • Children
  • Genetic predisposition
  • Risk
  • Second neoplasms

ASJC Scopus subject areas

  • Cancer Research
  • Oncology

Cite this

Second Neoplasms in Pediatric Patients with Primary Central Nervous System Tumors : The St. Jude Children's Research Hospital Experience. / Broniscer, Alberto; Ke, Weiming; Fuller, Christine E.; Wu, Jianrong; Gajjar, Amar; Kun, Larry E.

In: Cancer, Vol. 100, No. 10, 15.05.2004, p. 2246-2252.

Research output: Contribution to journalReview article

Broniscer, Alberto ; Ke, Weiming ; Fuller, Christine E. ; Wu, Jianrong ; Gajjar, Amar ; Kun, Larry E. / Second Neoplasms in Pediatric Patients with Primary Central Nervous System Tumors : The St. Jude Children's Research Hospital Experience. In: Cancer. 2004 ; Vol. 100, No. 10. pp. 2246-2252.
@article{54a1b0557ee74cc49c176486829228f6,
title = "Second Neoplasms in Pediatric Patients with Primary Central Nervous System Tumors: The St. Jude Children's Research Hospital Experience",
abstract = "BACKGROUND. Details on second neoplasms (SNs) following pediatric central nervous system (CNS) tumors are scant, because of the rarity of such SNs. The goal of the current study was to investigate and characterize these rare SNs. METHODS. The authors reviewed clinical and treatment data on all institutional patients age < 22 years at diagnosis of a primary CNS tumor who developed any type of SN. Patients with neurofibromatosis type 1 were excluded. Cumulative incidence rates were estimated, and putative risk factors were analyzed. RESULTS. The SNs investigated in the current study included 10 gliomas (42{\%}), 5 meningiomas (21{\%}), 2 desmoid tumors, 2 myelodysplastic syndromes, 2 basal cell carcinomas, 1 leukemia, 1 malignant fibrous histiocytoma, and 1 thyroid carcinoma. Twenty-one patients had previously received radiotherapy, and 12 patients had received chemotherapy. The SN was related to a genetic cause in 7 patients (29{\%}). Eleven patients died of their SNs, including 8 patients with glioma and 2 patients with myelodysplastic syndromes. The estimated 15-year cumulative incidence rate for malignant SNs was 4{\%}. Children with choroid plexus tumors had an estimated 10-year cumulative incidence rate of 20.2{\%}; 2 of those patients had germline TP53 mutations. Age ≤ 2 years was a significant risk factor (P = 0.016) for development of an SN only when patients with genetic conditions were included in the analysis. No significant difference in the estimated cumulative incidence of SNs was found among patients who had received different types of therapy. CONCLUSIONS. The risk of lethal SNs after pediatric CNS tumors is small. Young patients and patients with choroid plexus tumors appear to have an increased risk of SNs that is associated with genetic factors.",
keywords = "Brain, Central nervous system, Children, Genetic predisposition, Risk, Second neoplasms",
author = "Alberto Broniscer and Weiming Ke and Fuller, {Christine E.} and Jianrong Wu and Amar Gajjar and Kun, {Larry E.}",
year = "2004",
month = "5",
day = "15",
doi = "10.1002/cncr.20253",
language = "English (US)",
volume = "100",
pages = "2246--2252",
journal = "Cancer",
issn = "0008-543X",
publisher = "John Wiley and Sons Inc.",
number = "10",

}

TY - JOUR

T1 - Second Neoplasms in Pediatric Patients with Primary Central Nervous System Tumors

T2 - The St. Jude Children's Research Hospital Experience

AU - Broniscer, Alberto

AU - Ke, Weiming

AU - Fuller, Christine E.

AU - Wu, Jianrong

AU - Gajjar, Amar

AU - Kun, Larry E.

PY - 2004/5/15

Y1 - 2004/5/15

N2 - BACKGROUND. Details on second neoplasms (SNs) following pediatric central nervous system (CNS) tumors are scant, because of the rarity of such SNs. The goal of the current study was to investigate and characterize these rare SNs. METHODS. The authors reviewed clinical and treatment data on all institutional patients age < 22 years at diagnosis of a primary CNS tumor who developed any type of SN. Patients with neurofibromatosis type 1 were excluded. Cumulative incidence rates were estimated, and putative risk factors were analyzed. RESULTS. The SNs investigated in the current study included 10 gliomas (42%), 5 meningiomas (21%), 2 desmoid tumors, 2 myelodysplastic syndromes, 2 basal cell carcinomas, 1 leukemia, 1 malignant fibrous histiocytoma, and 1 thyroid carcinoma. Twenty-one patients had previously received radiotherapy, and 12 patients had received chemotherapy. The SN was related to a genetic cause in 7 patients (29%). Eleven patients died of their SNs, including 8 patients with glioma and 2 patients with myelodysplastic syndromes. The estimated 15-year cumulative incidence rate for malignant SNs was 4%. Children with choroid plexus tumors had an estimated 10-year cumulative incidence rate of 20.2%; 2 of those patients had germline TP53 mutations. Age ≤ 2 years was a significant risk factor (P = 0.016) for development of an SN only when patients with genetic conditions were included in the analysis. No significant difference in the estimated cumulative incidence of SNs was found among patients who had received different types of therapy. CONCLUSIONS. The risk of lethal SNs after pediatric CNS tumors is small. Young patients and patients with choroid plexus tumors appear to have an increased risk of SNs that is associated with genetic factors.

AB - BACKGROUND. Details on second neoplasms (SNs) following pediatric central nervous system (CNS) tumors are scant, because of the rarity of such SNs. The goal of the current study was to investigate and characterize these rare SNs. METHODS. The authors reviewed clinical and treatment data on all institutional patients age < 22 years at diagnosis of a primary CNS tumor who developed any type of SN. Patients with neurofibromatosis type 1 were excluded. Cumulative incidence rates were estimated, and putative risk factors were analyzed. RESULTS. The SNs investigated in the current study included 10 gliomas (42%), 5 meningiomas (21%), 2 desmoid tumors, 2 myelodysplastic syndromes, 2 basal cell carcinomas, 1 leukemia, 1 malignant fibrous histiocytoma, and 1 thyroid carcinoma. Twenty-one patients had previously received radiotherapy, and 12 patients had received chemotherapy. The SN was related to a genetic cause in 7 patients (29%). Eleven patients died of their SNs, including 8 patients with glioma and 2 patients with myelodysplastic syndromes. The estimated 15-year cumulative incidence rate for malignant SNs was 4%. Children with choroid plexus tumors had an estimated 10-year cumulative incidence rate of 20.2%; 2 of those patients had germline TP53 mutations. Age ≤ 2 years was a significant risk factor (P = 0.016) for development of an SN only when patients with genetic conditions were included in the analysis. No significant difference in the estimated cumulative incidence of SNs was found among patients who had received different types of therapy. CONCLUSIONS. The risk of lethal SNs after pediatric CNS tumors is small. Young patients and patients with choroid plexus tumors appear to have an increased risk of SNs that is associated with genetic factors.

KW - Brain

KW - Central nervous system

KW - Children

KW - Genetic predisposition

KW - Risk

KW - Second neoplasms

UR - http://www.scopus.com/inward/record.url?scp=2342644064&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=2342644064&partnerID=8YFLogxK

U2 - 10.1002/cncr.20253

DO - 10.1002/cncr.20253

M3 - Review article

C2 - 15139071

AN - SCOPUS:2342644064

VL - 100

SP - 2246

EP - 2252

JO - Cancer

JF - Cancer

SN - 0008-543X

IS - 10

ER -