Duan Yongheng, Lin Sheng, Xie Lichun, Zheng Kaifeng, Chen Shiguo, Song Hui, Zeng Xuchun, Gu Xueying, Wang Heyun, Zhang Linghua, Shao Hao, Hong Wenxu, Zhang Lijie, Duan Shan
Laboratory of Medical Genetics, Center for Birth Defect Research and Prevention, Shenzhen Research Institute of Population and Family Planning, Shenzhen City, People's Republic of China.
College of Pharmacy, Nankai University, Tianjin City, People's Republic of China.
Genet Mol Biol. 2017 Jul-Sep;40(3):591-596. doi: 10.1590/1678-4685-GMB-2016-0249. Epub 2017 Aug 31.
X-linked intellectual disability (XLID) has been associated with various genes. Diagnosis of XLID, especially for non-syndromic ones (NS-XLID), is often hampered by the heterogeneity of this disease. Here we report the case of a Chinese family in which three males suffer from intellectual disability (ID). The three patients shared the same phenotype: no typical clinical manifestation other than IQ score ≤ 70. For a genetic diagnosis for this family we carried out whole exome sequencing on the proband, and validated 16 variants of interest in the genomic DNA of all the family members. A missense mutation (c.710G > T), which mapped to exon 6 of the Rab GDP-Dissociation Inhibitor 1 (GDI1) gene, was found segregating with the ID phenotype, and this mutation changes the 237th position in the guanosine diphosphate dissociation inhibitor (GDI) protein from glycine to valine (p. Gly237Val). Through molecular dynamics simulations we found that this substitution results in a conformational change of GDI, possibly affecting the Rab-binding capacity of this protein. In conclusion, our study identified a novel GDI1 mutation that is possibly NS-XLID causative, and showed that whole exome sequencing provides advantages for detecting novel ID-associated variants and can greatly facilitate the genetic diagnosis of the disease.
X连锁智力障碍(XLID)与多种基因相关。XLID的诊断,尤其是非综合征型(NS-XLID)的诊断,常常受到该疾病异质性的阻碍。在此,我们报告一个中国家庭的病例,该家庭中有三名男性患有智力障碍(ID)。这三名患者具有相同的表型:除智商得分≤70外无典型临床表现。为了对这个家庭进行基因诊断,我们对先证者进行了全外显子组测序,并在所有家庭成员的基因组DNA中验证了16个感兴趣的变异。发现一个错义突变(c.710G>T),该突变定位于Rab GDP解离抑制剂1(GDI1)基因的第6外显子,与ID表型共分离,该突变使鸟苷二磷酸解离抑制剂(GDI)蛋白的第237位氨基酸由甘氨酸变为缬氨酸(p.Gly237Val)。通过分子动力学模拟,我们发现这种取代导致GDI构象改变,可能影响该蛋白与Rab的结合能力。总之,我们的研究鉴定出一种可能导致NS-XLID的新型GDI1突变,并表明全外显子组测序在检测与ID相关的新型变异方面具有优势,能够极大地促进该疾病的基因诊断。