Engberg Hedvig, Butwicka Agnieszka, Nordenström Anna, Hirschberg Angelica Lindén, Falhammar Henrik, Lichtenstein Paul, Nordenskjöld Agneta, Frisén Louise, Landén Mikael
Department of Clinical Neuroscience, Karolinska Institutet, Stockholm, Sweden; Child and Adolescent Psychiatry Research Center, Stockholm, Sweden.
Department of Medical Epidemiology and Biostatistics, Karolinska Institutet, Stockholm, Sweden; Department of Child Psychiatry, Medical University of Warsaw, Warsaw, Poland.
Psychoneuroendocrinology. 2015 Oct;60:195-205. doi: 10.1016/j.psyneuen.2015.06.017. Epub 2015 Jun 26.
Congenital adrenal hyperplasia (CAH) is a chronic condition and individuals are exposed to elevated androgen levels in utero as a result of the endogenous cortisol deficiency. Prenatal androgen exposure has been suggested to influence mental health, but population based studies on psychiatric morbidity among girls and women with CAH are lacking. Therefore, we performed a cohort study based on Swedish nationwide registers linked with the national CAH register. Girls and women with CAH due to 21-hydroxylase deficiency (n = 335) born between January 1915 and January 2010 were compared with aged-matched female (n = 33500) and male controls (n = 33500). Analyses were stratified by phenotype [salt wasting (SW), simple virilizing (SV), and non-classical type (NC)] and by CYP21A2 genotype subgroups (null, I2splice, I172N, and P30L). Results are presented as estimated risks (OR, 95%CI) of psychiatric disorders among girls and women with CAH compared with age-matched controls. Any psychiatric diagnoses were more common in CAH females compared with female and male population controls [1.9 (1.4-2.5), and 2.2 (1.7-2.9)]. In particular, the risk of alcohol misuse was increased compared with female and male population controls [2.8 (1.7-4.7) and 2.1 (1.2-3.5)], and appeared most common among the girls and women with the most severe null genotype [6.7 (2.6-17.8)]. The risk of stress and adjustment disorders was doubled compared with female population controls [2.1 (1.3-3.6)]. Girls and women with CAH have an increased risk of psychiatric disorders in general and substance use disorders in particular compared with unexposed females, with the highest risk among those with the most severe genotype. Prenatal androgen exposure and deficient endogenous cortisol and/or adrenaline production may provide explanations for these findings, but other factors related to CAH cannot be excluded.
先天性肾上腺皮质增生症(CAH)是一种慢性疾病,由于内源性皮质醇缺乏,个体在子宫内就暴露于升高的雄激素水平。产前雄激素暴露被认为会影响心理健康,但缺乏基于人群的关于CAH女孩和女性精神疾病发病率的研究。因此,我们基于瑞典全国性登记册与国家CAH登记册的关联进行了一项队列研究。将1915年1月至2010年1月期间出生的因21 - 羟化酶缺乏导致CAH的女孩和女性(n = 335)与年龄匹配的女性对照组(n = 33500)和男性对照组(n = 33500)进行比较。分析按表型[失盐型(SW)、单纯男性化型(SV)和非经典型(NC)]以及CYP21A2基因亚组(无效型、I2剪接型、I172N型和P30L型)进行分层。结果以CAH女孩和女性与年龄匹配对照组相比患精神疾病的估计风险(OR,95%CI)呈现。与女性和男性人群对照组相比,CAH女性中任何精神疾病诊断更为常见[1.9(1.4 - 2.5)和2.2(1.7 - 2.9)]。特别是,与女性和男性人群对照组相比,酒精滥用风险增加[2.8(1.7 - 4.7)和2.1(1.2 - 3.5)],并且在具有最严重无效基因型的女孩和女性中最为常见[6.7(2.6 - 17.8)]。与女性人群对照组相比,应激和适应障碍风险翻倍[2.1(1.3 - 3.6)]。与未暴露的女性相比,CAH女孩和女性总体上患精神疾病的风险增加,尤其是物质使用障碍风险增加,在基因型最严重的人群中风险最高。产前雄激素暴露以及内源性皮质醇和/或肾上腺素分泌不足可能为这些发现提供解释,但与CAH相关的其他因素也不能排除。