title: Reproductive problems and birth defects in survivors of Wilms' tumor and their relatives.
abstract: In a retrospective cohort study of 47 Wilms' tumor survivors and their 77 sibling controls, female survivors had a fourfold excess risk (risk ratio, 4.1; 95% confidence interval, 1.7-10.1) for any adverse livebirth outcome, including birth defects, compared with their sibling controls. Wives of male survivors had no apparent excess risk for problem pregnancies. The families had a number of severe reproductive problems and major birth defects, such as primary amenorrhea in two survivors, bicornuate uterus in two survivors and one control, and mental retardation in one male survivor and a male control. The son of a female survivor died after bilateral Wilms' tumors. Birth defects in the offspring of female survivors are compatible either with intrauterine constraint, possibly due to radiation-induced fibrosis or with the complex of malformations associated with Wilms' tumor. Female survivors of Wilms' tumor appear to be at increased risk for a variety of reproductive problems, from sterility to fetal loss, early delivery, and birth defects in offspring. Furthermore, relatives of survivors of Wilms' tumor may be at risk of having associated birth defects, with clinically significant consequences.
authors.affiliation: Clinical Epidemiology Branch, National Cancer Institute, Bethesda, MD 20892.
authors.forename: J
authors.initials: J
authors.lastname: Byrne
authors.forename: J J
authors.initials: JJ
authors.lastname: Mulvihill
authors.forename: R R
authors.initials: RR
authors.lastname: Connelly
authors.forename: D A
authors.initials: DA
authors.lastname: Austin
authors.forename: G E
authors.initials: GE
authors.lastname: Holmes
authors.forename: F F
authors.initials: FF
authors.lastname: Holmes
authors.forename: H B
authors.initials: HB
authors.lastname: Latourette
authors.forename: J W
authors.initials: JW
authors.lastname: Meigs
authors.forename: L C
authors.initials: LC
authors.lastname: Strong
authors.forename: M H
authors.initials: MH
authors.lastname: Myers
mesh_terms: D000293:Adolescent; D000328:Adult; D002648:Child; D000013:Congenital Abnormalities / Q000453:epidemiology* / Q000235:genetics; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D007680:Kidney Neoplasms / Q000235:genetics / Q000401:mortality*; D008297:Male; D008599:Menstruation Disturbances / Q000453:epidemiology / Q000235:genetics; D011247:Pregnancy; D011256:Pregnancy Outcome; D012098:Reproduction; D012189:Retrospective Studies; D012307:Risk Factors; D009396:Wilms Tumor / Q000235:genetics / Q000401:mortality*