Pang Xiuhong, Luo Huajie, Chai Yongchuan, Wang Xiaowen, Sun Lianhua, He Longxia, Chen Penghui, Wu Hao, Yang Tao
Department of Otolaryngology-Head and Neck Surgery, Xinhua Hospital, Shanghai Jiaotong University School of Medicine, Shanghai, China; Ear Institute, Shanghai Jiaotong University School of Medicine, Shanghai, China; Shanghai Key Laboratory of Translational Medicine on Ear and Nose Diseases, Shanghai, China; Department of Otorhinolaryngology-Head and Neck Surgery, Taizhou People's Hospital, Jiangsu Province, China.
Department of Otolaryngology, Renji hospital, Shanghai Jiaotong University School of Medicine, Shanghai, China.
PLoS One. 2015 Mar 27;10(3):e0120816. doi: 10.1371/journal.pone.0120816. eCollection 2015.
Microdeletions in chromosome 17q22, where the NOG gene resides, have been reported leading to the NOG-related symphalangism spectrum disorder (NOG-SSD), intellectual disability and other developmental abnormalities. In this study we reported a dominant Chinese Han family segregating with typical NOG-SSD symptoms including proximal symphalangism, conductive hearing loss, amblyopia and strabismus, but not intellectual disability. Sanger sequencing identified no pathogenic mutation in the coding regions of candidate genes NOG, GDF5 and FGF9. SNP genotyping in the genomic region surrounding NOG identified loss of heterozygosity in the affected family members. By array comparative genomic hybridization and quantitative real-time polymerase chain reaction, we identified and mapped the breakpoints of a novel 1.6-Mb microdeletion in chromosome 17q22 that included NOG and twelve other genes. It is the first microdeletion reported in chromosome 17q22 that is associated with NOG-SSD only but not with intellectual disability. Our results may help identifying the dosage sensitive genes for intellectual disability and other developmental abnormalities in chromosome 17q22. Our study also suggested that genomic deletions in chromosome 17q22 should be screened in the NOG-SSD patients in which no pathogenic mutation is identified by conventional sequencing methods.
17号染色体q22区域存在微小缺失,NOG基因就位于该区域,据报道这种缺失会导致与NOG相关的并指畸形谱系障碍(NOG-SSD)、智力残疾和其他发育异常。在本研究中,我们报告了一个显性遗传的中国汉族家系,该家系具有典型的NOG-SSD症状,包括近端并指畸形、传导性听力损失、弱视和斜视,但无智力残疾。桑格测序未在候选基因NOG、GDF5和FGF9的编码区发现致病突变。对NOG周围基因组区域进行单核苷酸多态性基因分型,发现患病家庭成员存在杂合性缺失。通过阵列比较基因组杂交和定量实时聚合酶链反应,我们鉴定并定位了17号染色体q22区域一个新的1.6兆碱基微小缺失的断点,该缺失包含NOG和其他12个基因。这是首次报道的17号染色体q22区域仅与NOG-SSD相关而与智力残疾无关的微小缺失。我们的结果可能有助于确定17号染色体q22区域中对智力残疾和其他发育异常具有剂量敏感性的基因。我们的研究还表明,对于那些通过传统测序方法未发现致病突变的NOG-SSD患者,应筛查17号染色体q22区域的基因组缺失。