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DOI 10.1172/jci183758

Медицинская библиотека · The Journal of clinical investigation · 2024

MGA loss-of-function variants cause premature ovarian insufficiency.

Tang Shuyan, Guo Ting, Song Chengcheng, Wang Lingbo, Zhang Jun, Rajkovic Aleksandar, Lin Xiaoqi, Chen Shiling, Liu Yujun, Tian Weidong, Wu Bangguo, Wang Shixuan, Wang Wenwen, Lai Yunhui, Wang Ao, Xu Shuhua, Jin Li, Ke Hanni, Zhao Shidou, Li Yan, Qin Yingying, Zhang Feng, Chen Zi-Jiang

СтатьяПоддержка исследования: вне правительства СШАПоддержка NIH: внешняя программаПеревод в очереди

Аннотация

Although premature ovarian insufficiency (POI), a common cause of female infertility and subfertility, has a well-established hereditary component, the genetic factors currently implicated in POI account for only a limited proportion of cases. Here, using an exome-wide, gene-based case-control analysis in a discovery cohort comprising 1,027 POI cases and 2,733 ethnically matched women controls from China, we found that heterozygous loss-of-function (LoF) variants of MAX dimerization protein (MGA) were significantly enriched in the discovery cohort, accounting for 2.6% of POI cases, while no MGA LoF variants were found in the matched control females. Further exome screening was conducted in 4 additional POI cohorts (2 from China and 2 from the United States) for replication studies, and we identified heterozygous MGA LoF variants in 1.0%, 1.4%, 1.0%, and 1.0% of POI cases, respectively. Overall, a total of 37 distinct heterozygous MGA LoF variants were discovered in 38 POI cases, accounting for approximately 2.0% of the total 1,910 POI cases analyzed in this study. Accordingly, Mga+/- female mice were subfertile, exhibiting shorter reproductive lifespan and decreased follicle number compared with WT, mimicking the observed phenotype in humans. Our findings highlight the essential role of MGA deficiency for impaired female reproductive ability.

Для цитирования: Tang Shuyan, Guo Ting, Song Chengcheng, Wang Lingbo, Zhang Jun, Rajkovic Aleksandar, Lin Xiaoqi, Chen Shiling, Liu Yujun, Tian Weidong, Wu Bangguo, Wang Shixuan, Wang Wenwen, Lai Yunhui, Wang Ao, Xu Shuhua, Jin Li, Ke Hanni, Zhao Shidou, Li Yan, Qin Yingying, Zhang Feng, Chen Zi-Jiang. MGA loss-of-function variants cause premature ovarian insufficiency.. The Journal of clinical investigation. 2024. DOI: 10.1172/jci183758.

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