Andonova Silvia, Robeva Ralitsa, Sirakov Milko, Mainhard Karela, Tomova Analia, Ledig Susanne, Kumanov Philip, Savov Alexey
National Genetic Laboratory - UHOG x2018;Maichin dom', Sofia, Bulgaria.
Sex Dev. 2015;9(6):333-7. doi: 10.1159/000443807. Epub 2016 Feb 13.
46,XY complete gonadal dysgenesis (CGD) is a disorder of sexual development that can result from different mutations in genes associated with sex determination. Patients are phenotypically females, and the disease is often diagnosed in late adolescence because of delayed puberty. Here, we present the clinical and molecular data of a 46,XY female CGD patient with gonadoblastoma with dysgerminoma and incidentally found inherited thrombophilia. The clinical significance of the described de novo SRY gene mutation c.325T>C (p.F109L) is discussed. This case report supports the critical role of the HGM domain in the SRY gene and the need of a multidisciplinary approach for CGD patients.
46,XY完全性性腺发育不全(CGD)是一种性发育障碍,可由与性别决定相关基因的不同突变引起。患者表型为女性,由于青春期延迟,该病常在青春期末期被诊断出来。在此,我们报告一例46,XY女性CGD患者的临床和分子数据,该患者患有性腺母细胞瘤伴无性细胞瘤,且偶然发现遗传性血栓形成倾向。讨论了所描述的新发SRY基因突变c.325T>C(p.F109L)的临床意义。本病例报告支持HGM结构域在SRY基因中的关键作用以及CGD患者采取多学科方法的必要性。