Ho Chi Minh City Hospital of Dermato-Venereology, Ho Chi Minh City, Vietnam.
Center for Molecular Biomedicine, University of Medicine and Pharmacy at Ho Chi Minh City, Ho Chi Minh City, Vietnam.
Am J Med Genet A. 2022 Jan;188(1):377-381. doi: 10.1002/ajmg.a.62477. Epub 2021 Aug 28.
Cantú syndrome (CS) is an extremely rare autosomal dominant hereditary disease characterized by congenital hypertrichosis, distinct coarse facial features, cardiac defects, and other abnormalities in the skeletal and neurological systems. At present, cases with pathognomonic clinical manifestations are increasingly confirmed by genetic analysis. Two causative genes for CS are the well-known ABCC9 and the more rarely reported KCNJ8. Here, we report three Vietnamese children with CS, confirmed through genetic testing, presenting de novo ABCC9 mutations. The patients shared some common clinical manifestations, including congenital hypertrichosis, distinctive facial features, and a history of polyhydramnios during pregnancy. Concerning the various cardiac and neurological problems in the lifetime of patients with CS, an accurate diagnosis and appropriate management, especially genetic counseling, should be clinically applied in CS. Thus, our findings might modestly contribute to the global CS data, providing practical insights into CS manifestations.
坎图综合征(CS)是一种极为罕见的常染色体显性遗传性疾病,其特征为先天性多毛症、明显的粗糙面容、心脏缺陷以及骨骼和神经系统的其他异常。目前,通过基因分析越来越多地证实了具有特征性临床表现的病例。CS 的两个致病基因是众所周知的 ABCC9 和报道较少的 KCNJ8。在这里,我们报告了三例通过基因检测确诊的越南 CS 患儿,均存在新发 ABCC9 突变。这些患者具有一些共同的临床表现,包括先天性多毛症、独特的面容以及妊娠期间羊水过多的病史。鉴于 CS 患者一生中存在各种心脏和神经系统问题,临床上应进行准确的诊断和适当的管理,尤其是遗传咨询。因此,我们的发现可能会对全球 CS 数据略有贡献,为 CS 表现提供实用的见解。