Congenital abnormalities and hepatoblastoma: A report from the Children's Oncology Group (COG)

Logan G Spector, Susan E. Puumala, Michael K Georgieff, Mark D. Krailo, Gail Tomlinson

Research output: Book/ReportBook

Abstract

Hepatoblastoma (HB) is a rare pediatric tumor which comprises the majority of liver cancer in children. Previous research has established that Beckwith-Wiedemann Syndrome (BWS) and Familial Adenomatous Polyposis (FAP) predispose to HB, but information on its association with isolated congenital abnormalities is sparse. We examined data from a case-control study of HB conducted through the Children's Oncology Group (COG). Cases were diagnosed by COG institutions during 2000-2008 and controls were recruited from state birth registries, frequency matched for sex, region, year of birth, and low birth weight (a strong risk factor for HB). Data on congenital abnormalities among subjects and covariates were obtained by maternal telephone interview. There were 359 cases and 372 controls included in this analysis, which comprises approximately 90% of the final sample. Odds ratios (OR) and 95% confidence intervals (CI) describing the association between congenital abnormalities with HB, adjusted for birth weight and sex, were calculated using unconditional logistic regression. Twelve cases were reported to have BWS and 4 to have FAP, while no controls had either condition; further analyses excluded abnormalities among children with these syndromes to better examine isolated defects. There was a significant association of HB with kidney, bladder, or sex organ abnormalities (OR = 4.22; 95% CI: 1.57-11.29) which appeared to be specific to kidney/bladder defects (OR = 5.24; 95% CI: 1.40-19.59) but not those of sex organs (OR = 1.58; 95% CI: 0.45-5.55). Elevated but non-significant ORs were found for spina bifida or other spinal defects (OR = 2.39; 95% CI: 0.40-14.07), large or multiple birthmarks (OR = 1.63; 95% CI: 0.97-2.76). No significant associations of cleft lip or palate, Down syndrome or other numerical chromosomal anomalies, microcephaly, or rib abnormalities, although
Original languageEnglish (US)
Volume70
StatePublished - 2010

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Hepatoblastoma
Odds Ratio
Confidence Intervals
Beckwith-Wiedemann Syndrome
Adenomatous Polyposis Coli
Urinary Bladder
Parturition
Kidney
Microcephaly
Spinal Dysraphism
Cleft Lip
Cleft Palate
Low Birth Weight Infant
Ribs
Liver Neoplasms
Down Syndrome
Birth Weight
Registries
Case-Control Studies
Logistic Models

Keywords

  • *cancer research
  • *child
  • *congenital disorder
  • *hepatoblastoma
  • *human
  • *oncology
  • Beckwith Wiedemann syndrome
  • birth weight
  • bladder
  • case control study
  • chromosome aberration
  • cleft lip
  • confidence interval
  • Down syndrome
  • familial colon polyposis
  • kidney
  • liver cancer
  • logistic regression analysis
  • low birth weight
  • microcephaly
  • neoplasm
  • palate
  • register
  • rib malformation
  • risk
  • risk factor
  • skin hemangioma
  • spinal dysraphism
  • telephone interview

Cite this

Congenital abnormalities and hepatoblastoma: A report from the Children's Oncology Group (COG). / Spector, Logan G; Puumala, Susan E.; Georgieff, Michael K; Krailo, Mark D.; Tomlinson, Gail.

2010.

Research output: Book/ReportBook

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title = "Congenital abnormalities and hepatoblastoma: A report from the Children's Oncology Group (COG)",
abstract = "Hepatoblastoma (HB) is a rare pediatric tumor which comprises the majority of liver cancer in children. Previous research has established that Beckwith-Wiedemann Syndrome (BWS) and Familial Adenomatous Polyposis (FAP) predispose to HB, but information on its association with isolated congenital abnormalities is sparse. We examined data from a case-control study of HB conducted through the Children's Oncology Group (COG). Cases were diagnosed by COG institutions during 2000-2008 and controls were recruited from state birth registries, frequency matched for sex, region, year of birth, and low birth weight (a strong risk factor for HB). Data on congenital abnormalities among subjects and covariates were obtained by maternal telephone interview. There were 359 cases and 372 controls included in this analysis, which comprises approximately 90{\%} of the final sample. Odds ratios (OR) and 95{\%} confidence intervals (CI) describing the association between congenital abnormalities with HB, adjusted for birth weight and sex, were calculated using unconditional logistic regression. Twelve cases were reported to have BWS and 4 to have FAP, while no controls had either condition; further analyses excluded abnormalities among children with these syndromes to better examine isolated defects. There was a significant association of HB with kidney, bladder, or sex organ abnormalities (OR = 4.22; 95{\%} CI: 1.57-11.29) which appeared to be specific to kidney/bladder defects (OR = 5.24; 95{\%} CI: 1.40-19.59) but not those of sex organs (OR = 1.58; 95{\%} CI: 0.45-5.55). Elevated but non-significant ORs were found for spina bifida or other spinal defects (OR = 2.39; 95{\%} CI: 0.40-14.07), large or multiple birthmarks (OR = 1.63; 95{\%} CI: 0.97-2.76). No significant associations of cleft lip or palate, Down syndrome or other numerical chromosomal anomalies, microcephaly, or rib abnormalities, although",
keywords = "*cancer research, *child, *congenital disorder, *hepatoblastoma, *human, *oncology, Beckwith Wiedemann syndrome, birth weight, bladder, case control study, chromosome aberration, cleft lip, confidence interval, Down syndrome, familial colon polyposis, kidney, liver cancer, logistic regression analysis, low birth weight, microcephaly, neoplasm, palate, register, rib malformation, risk, risk factor, skin hemangioma, spinal dysraphism, telephone interview",
author = "Spector, {Logan G} and Puumala, {Susan E.} and Georgieff, {Michael K} and Krailo, {Mark D.} and Gail Tomlinson",
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T1 - Congenital abnormalities and hepatoblastoma: A report from the Children's Oncology Group (COG)

AU - Spector, Logan G

AU - Puumala, Susan E.

AU - Georgieff, Michael K

AU - Krailo, Mark D.

AU - Tomlinson, Gail

PY - 2010

Y1 - 2010

N2 - Hepatoblastoma (HB) is a rare pediatric tumor which comprises the majority of liver cancer in children. Previous research has established that Beckwith-Wiedemann Syndrome (BWS) and Familial Adenomatous Polyposis (FAP) predispose to HB, but information on its association with isolated congenital abnormalities is sparse. We examined data from a case-control study of HB conducted through the Children's Oncology Group (COG). Cases were diagnosed by COG institutions during 2000-2008 and controls were recruited from state birth registries, frequency matched for sex, region, year of birth, and low birth weight (a strong risk factor for HB). Data on congenital abnormalities among subjects and covariates were obtained by maternal telephone interview. There were 359 cases and 372 controls included in this analysis, which comprises approximately 90% of the final sample. Odds ratios (OR) and 95% confidence intervals (CI) describing the association between congenital abnormalities with HB, adjusted for birth weight and sex, were calculated using unconditional logistic regression. Twelve cases were reported to have BWS and 4 to have FAP, while no controls had either condition; further analyses excluded abnormalities among children with these syndromes to better examine isolated defects. There was a significant association of HB with kidney, bladder, or sex organ abnormalities (OR = 4.22; 95% CI: 1.57-11.29) which appeared to be specific to kidney/bladder defects (OR = 5.24; 95% CI: 1.40-19.59) but not those of sex organs (OR = 1.58; 95% CI: 0.45-5.55). Elevated but non-significant ORs were found for spina bifida or other spinal defects (OR = 2.39; 95% CI: 0.40-14.07), large or multiple birthmarks (OR = 1.63; 95% CI: 0.97-2.76). No significant associations of cleft lip or palate, Down syndrome or other numerical chromosomal anomalies, microcephaly, or rib abnormalities, although

AB - Hepatoblastoma (HB) is a rare pediatric tumor which comprises the majority of liver cancer in children. Previous research has established that Beckwith-Wiedemann Syndrome (BWS) and Familial Adenomatous Polyposis (FAP) predispose to HB, but information on its association with isolated congenital abnormalities is sparse. We examined data from a case-control study of HB conducted through the Children's Oncology Group (COG). Cases were diagnosed by COG institutions during 2000-2008 and controls were recruited from state birth registries, frequency matched for sex, region, year of birth, and low birth weight (a strong risk factor for HB). Data on congenital abnormalities among subjects and covariates were obtained by maternal telephone interview. There were 359 cases and 372 controls included in this analysis, which comprises approximately 90% of the final sample. Odds ratios (OR) and 95% confidence intervals (CI) describing the association between congenital abnormalities with HB, adjusted for birth weight and sex, were calculated using unconditional logistic regression. Twelve cases were reported to have BWS and 4 to have FAP, while no controls had either condition; further analyses excluded abnormalities among children with these syndromes to better examine isolated defects. There was a significant association of HB with kidney, bladder, or sex organ abnormalities (OR = 4.22; 95% CI: 1.57-11.29) which appeared to be specific to kidney/bladder defects (OR = 5.24; 95% CI: 1.40-19.59) but not those of sex organs (OR = 1.58; 95% CI: 0.45-5.55). Elevated but non-significant ORs were found for spina bifida or other spinal defects (OR = 2.39; 95% CI: 0.40-14.07), large or multiple birthmarks (OR = 1.63; 95% CI: 0.97-2.76). No significant associations of cleft lip or palate, Down syndrome or other numerical chromosomal anomalies, microcephaly, or rib abnormalities, although

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KW - child

KW - congenital disorder

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KW - human

KW - oncology

KW - Beckwith Wiedemann syndrome

KW - birth weight

KW - bladder

KW - case control study

KW - chromosome aberration

KW - cleft lip

KW - confidence interval

KW - Down syndrome

KW - familial colon polyposis

KW - kidney

KW - liver cancer

KW - logistic regression analysis

KW - low birth weight

KW - microcephaly

KW - neoplasm

KW - palate

KW - register

KW - rib malformation

KW - risk

KW - risk factor

KW - skin hemangioma

KW - spinal dysraphism

KW - telephone interview

M3 - Book

VL - 70

BT - Congenital abnormalities and hepatoblastoma: A report from the Children's Oncology Group (COG)

ER -