Childhood cancer in children with congenital anomalies in Oklahoma, 1997 to 2009

Abstract

Background Data-linkage studies have reported an association between congenital anomalies and childhood cancer. However, few studies have focused on the differences in the effect of congenital anomalies on cancer as a function of attained age. We aimed to examine associations between anomalies and childhood cancer as a function of attained age among children born in Oklahoma. Methods Data were obtained from the Oklahoma State Department of Health from 1997-2009 (n=591,235). We linked Vital Statistics records for singleton deliveries to the Oklahoma Birth Defects Registry and the Oklahoma Central Cancer Registry using name and birth date. In order to assess the relation between anomalies and childhood cancer, we used Cox regression analysis allowing for a non-proportional hazards for anomalies as a function of age. Results There were 23,368 (4.0%) children with anomalies and 531 (0.1%) children with cancer. When considering 3-year age intervals, we detected an increased hazard of any childhood cancer in children with anomalies compared to those without anomalies before one year of age (HR: 14.1, 95% CI: 8.3, 23.7) and at three years of age (HR: 2.3, 95% CI: 1.6, 3.2). The increased hazard declined with increasing time since birth, with the effect diminished by six years of age. Conclusions Our results were consistent with previous studies indicating an increased rate of childhood cancer among children with anomalies at younger ages. Furthermore, our study added a methodological refinement of assessing the effect of anomalies as a function of attained age.

Childhood cancer risk in those with chromosomal and non-chromosomal congenital anomalies in Washington State: 1984-2013

“…When only non-chromosomal anomalies were considered, the risk estimates generally remained increased, supporting results of other studies indicating an association with childhood cancer diagnosed at different ages, but with a slight decrease in risk with attained age. [5] We also observed increased cancer risk in relation to increasing number of anomalies present, which is consistent with one earlier report. [18] Also consistent with other population-based data linkage studies that examined non-chromosomal anomalies, we observed increased risks of selected cancer types.…”

“…[5–7] Our overall effect estimate for the risk of cancer among children with non-chromosomal anomalies (OR = 1.35) was only slightly attenuated compared to those reported by Janitz et al (HR = 2.50)[5], Botto et al (incidence rate ratio [IRR] = 2.00),[7] and Fisher et al (HR = 1.58).…”

“…Our study lends to the growing body of evidence that non-chromosomal anomalies are also associated with childhood cancer risk. [2, 5–7] Additionally, our results indicate that an increasing number of non-chromosomal anomalies was more strongly associated with increased cancer risk compared to those children with only one non-chromosomal anomaly (OR for three or more anomalies: 3.11, 95% CI: 1.54–6.11 vs. OR for one anomaly: 1.29, 95% CI: 1.12–1.49). This may suggest that children with previously unidentified multiple malformation syndromes (and no obvious chromosomal anomaly) may be at a significant risk of cancer.…”

“…We evaluated this association overall, and for cancer occurrence at different diagnosis age categories (<5, 5–9, 10–19 years) to be consistent with previous assessments. [5] Because congenital anomalies may be associated with infant birthweight or gestational age at delivery, which may also affect the risk of cancer occurrence, we conducted sub-analyses of our main exposures (any major anomaly, major non-chromosomal anomalies) restricted to children with normal birthweight (2500 - <4000g) and term gestation (37 weeks or greater). When numbers permitted, associations between specific anomalies and childhood cancer were examined.…”

“…[2, 3] Similarly, children with chromosome 13q14 deletion syndrome, characterized by dysmorphic facial features, have increased risk of retinoblastoma. [4] Four recent population-based registry linkage studies in the United States (U.S.)[2, 5–7] suggest that children with non-chromosomal anomalies may also be more likely to develop cancer compared to their unaffected contemporaries.…”