Takenouchi Toshiki, Miura Kiyokuni, Uehara Tomoko, Mizuno Seiji, Kosaki Kenjiro
Center for Medical Genetics, Keio University School of Medicine, Tokyo, Japan.
Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan.
Am J Med Genet A. 2016 Oct;170(10):2587-90. doi: 10.1002/ajmg.a.37761. Epub 2016 Jun 3.
A recent study of exome analyses in 109 patients with undiagnosed diseases included a 5-year-old girl with intellectual disability and multiple congenital anomalies, who had an apparently de novo frameshift mutation in SON. However, the combination of the truncating mutation in SON and the phenotype has not been reproduced until date, and it remains unclear if this combination represents a distinctive disease entity. Here we report an additional male with intellectual disability, congenital heart disease, distinctive facial features with curly hair and protruding ears, and long slender extremities, and hyperextensible joints. Exome analysis showed that he had the same de novo frameshift mutation in SON in a heterozygous state. Along with the first and original description of the apparently de novo truncating mutation in SON mentioned above, we have established that haploinsufficiency of SON causes a new recognizable syndrome of intellectual disability. SON is located within 21q22.11, a critical region for Braddock-Carey syndrome, which is characterized by congenital thrombocytopenia, intellectual disability, micrognathia, and a distinctive facies. Therefore, we suggest that the intellectual disability observed in Braddock-Carey syndrome could be accounted for by haploinsufficiency of SON. © 2016 Wiley Periodicals, Inc.
最近一项对109名未确诊疾病患者进行外显子组分析的研究,纳入了一名患有智力残疾和多种先天性异常的5岁女孩,她在SON基因中存在一个明显的新生移码突变。然而,SON基因中的截短突变与该表型的组合至今尚未得到重现,并且尚不清楚这种组合是否代表一种独特的疾病实体。在此,我们报告了另一名男性患者,他患有智力残疾、先天性心脏病、具有卷发和招风耳的独特面部特征、四肢细长以及关节过度伸展。外显子组分析显示,他在SON基因中存在相同的杂合状态新生移码突变。连同上述对SON基因中明显的新生截短突变的首次及原始描述,我们已确定SON基因单倍剂量不足会导致一种新的可识别的智力残疾综合征。SON基因位于21q22.11,这是布拉多克 - 凯里综合征的关键区域,其特征为先天性血小板减少、智力残疾、小颌畸形和独特面容。因此,我们认为布拉多克 - 凯里综合征中观察到的智力残疾可能是由SON基因单倍剂量不足所致。© 2016威利期刊公司