Department of Environmental Health, Harvard T.H. Chan School of Public Health, Boston, Massachusetts.
Maccabi Institute for Research and Innovation, Maccabi Healthcare Services, Tel Aviv, Israel.
Am J Epidemiol. 2018 Apr 1;187(4):656-663. doi: 10.1093/aje/kwx367.
Androgens have an extensive influence on brain development in regions of the brain that are relevant for autism spectrum disorder (ASD), yet their etiological involvement remains unclear. Hypospadias (abnormal positioning of the urethral opening) and cryptorchidism (undescended testes) are 2 relatively common male birth defects that are strongly associated with prenatal androgen deficiencies. Having either disorder is a proxy indicator of atypical gestational androgen exposure, yet the association between these disorders and autism has not been extensively studied. We analyzed male singleton live births (n = 224,598) occurring from January 1, 1999, through December 31, 2013, in a large Israeli health-care organization. Boys with autism, cryptorchidism, and hypospadias were identified via International Classification of Diseases, Ninth Revision, codes, with further verification of autism case status by review of medical records. In multivariable-adjusted analyses, the odds ratio for ASD among boys with either condition was 1.62 (95% confidence interval (CI): 1.44, 1.82). The odds ratio for boys with cryptorchidism only was 1.55 (95% CI: 1.34, 1.78), and that for boys with hypospadias only was 1.65 (95% CI: 1.38, 1.98). ASD risk was not elevated among unaffected brothers of hypospadias or cryptorchidism cases, despite familial aggregation of all 3 conditions, providing some indication for the possibility of pregnancy-specific risk factors driving the observed associations. Results suggest that in-utero hypoandrogenicity could play a role in ASD etiology.
雄激素对自闭症谱系障碍(ASD)相关脑区的大脑发育有广泛影响,但它们的病因学作用仍不清楚。尿道下裂(尿道开口位置异常)和隐睾(睾丸未降)是两种相对常见的男性出生缺陷,与产前雄激素缺乏密切相关。患有这两种疾病中的任何一种都是胎儿雄激素暴露异常的指标,但这些疾病与自闭症之间的关联尚未得到广泛研究。我们分析了 1999 年 1 月 1 日至 2013 年 12 月 31 日期间在一个大型以色列医疗保健组织中出生的 224598 名男性 singleton 活产儿。通过国际疾病分类,第九版代码,识别出患有自闭症、隐睾和尿道下裂的男孩,并通过审查病历进一步验证自闭症病例的状态。在多变量调整分析中,患有任何一种疾病的男孩患 ASD 的比值比为 1.62(95%置信区间(CI):1.44,1.82)。仅患有隐睾症的男孩的比值比为 1.55(95%置信区间(CI):1.34,1.78),仅患有尿道下裂症的男孩的比值比为 1.65(95%置信区间(CI):1.38,1.98)。尽管所有 3 种疾病都存在家族聚集性,但患有尿道下裂或隐睾症的未受影响兄弟的自闭症风险并未升高,这表明妊娠特异性危险因素可能导致了观察到的关联。结果表明,宫内低雄激素血症可能在 ASD 病因学中起作用。