Department of Medical Genetics, Ataturk University, Erzurum, Turkey.
Department of Medical Genetics, Istanbul University-Cerrahpasa, Istanbul, Turkey.
Am J Med Genet A. 2022 Sep;188(9):2815-2818. doi: 10.1002/ajmg.a.62861. Epub 2022 Jun 16.
Agenesis of Corpus Callosum, Cardiac, Ocular, and Genital Syndrome (ACOGS; OMIM #618929) is a rare genetic disorder characterized by global developmental delay, agenesis or hypoplasia of corpus callosum, craniofacial dysmorphism, ocular, cardiac, and genital anomalies. ACOGS is caused by variations in the CDH2 gene. Our patient had a novel finding besides the classical findings of ACOGS. To the best of our knowledge, only 14 patients with ACOGS have been reported. Here, we reported the fifteenth patient with ACOGS, having a novel de novo nonsense variant in the CDH2 gene, and the first patient from Turkey with a novel finding. Our patient was the first female to have a renal anomaly since only genital malformations were reported in male patients (cryptorchidism, micropenis) so far.
脑-心-眼-生殖器综合征(ACOGS;OMIM#618929)是一种罕见的遗传疾病,其特征为全面发育迟缓、胼胝体发育不全或发育不良、颅面畸形、眼部、心脏和生殖器异常。ACOGS 是由 CDH2 基因突变引起的。我们的患者除了具有 ACOGS 的典型表现外,还有一个新的发现。据我们所知,目前仅报道了 14 例 ACOGS 患者。在此,我们报告了第 15 例 ACOGS 患者,其 CDH2 基因存在新的从头错义变异,也是土耳其首例具有新发现的患者。我们的患者是首例出现肾脏异常的女性,因为迄今为止仅报道了男性患者的生殖器畸形(隐睾、小阴茎)。