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肾母细胞瘤幸存者及其亲属的生殖问题和出生缺陷。

Reproductive problems and birth defects in survivors of Wilms' tumor and their relatives.

作者信息

Byrne J, Mulvihill J J, Connelly R R, Austin D A, Holmes G E, Holmes F F, Latourette H B, Meigs J W, Strong L C, Myers M H

机构信息

Clinical Epidemiology Branch, National Cancer Institute, Bethesda, MD 20892.

出版信息

Med Pediatr Oncol. 1988;16(4):233-40. doi: 10.1002/mpo.2950160403.

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.

摘要

在一项对47名肾母细胞瘤幸存者及其77名同胞对照进行的回顾性队列研究中,与同胞对照相比,女性幸存者出现任何不良生育结局(包括出生缺陷)的风险高出四倍(风险比为4.1;95%置信区间为1.7 - 10.1)。男性幸存者的妻子在妊娠问题方面没有明显的额外风险。这些家庭存在许多严重的生殖问题和主要出生缺陷,例如两名幸存者出现原发性闭经,两名幸存者和一名对照者出现双子宫,一名男性幸存者和一名男性对照者出现智力发育迟缓。一名女性幸存者的儿子在患双侧肾母细胞瘤后死亡。女性幸存者后代的出生缺陷可能与宫内受限有关,这可能是由于辐射诱导的纤维化所致,也可能与肾母细胞瘤相关的畸形复合体有关。肾母细胞瘤女性幸存者出现各种生殖问题的风险似乎有所增加,从不育到胎儿丢失、早产以及后代出生缺陷。此外,肾母细胞瘤幸存者的亲属可能有出现相关出生缺陷的风险,并会产生具有临床意义的后果。

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