Department of Ophthalmology, The Third Affiliated Hospital, Sun Yat‑sen University, Guangzhou, Guangdong 510630, P.R. China.
Department of Infectious Diseases, The Third Affiliated Hospital, Sun Yat‑sen University, Guangzhou, Guangdong 510630, P.R. China.
Mol Med Rep. 2017 Aug;16(2):1753-1758. doi: 10.3892/mmr.2017.6824. Epub 2017 Jun 20.
The purpose of the current study was to identify novel mutations in the FRMD7 (FERM domain containing 7) gene and to characterize clinical features in Chinese patients with congenital motor nystagmus. For this purpose, 18 patients with congenital motor nystagmus were selected from the ocular genetic diseases bank of the Pediatric and Genetic Clinic of Zhongshan Ophthalmic Center (Guangdong, China). Direct sequencing was used to analyze the exons and adjacent introns of the FRMD7 gene. The heteroduplex‑single strand conformation polypeptide method was used to analyze 96 unrelated normal controls and gene‑screening positive patients. Slit lamp photography of the anterior segment, fundus photography, optical coherence tomography and electroretinogram were carried out to identify the clinical features of congenital motor nystagmus. The authors noted that in, 18 patients with congenital motor nystagmus, there were 7FRMD7 gene mutations (six new mutations). The screening rate was 38.89%, including c.41_43delAGA (p.13‑15delK); c.473T>A (p.I158N); c.605T>A (p.I202N); c.580G>T (p.A194S); c.811T>A (p.C271S); c.1493insA (p.Y498X); c.57+1G>A (slice mutation). There were no such mutations in the 96 normal controls. These results enriched the gene mutation spectrum of FRMD7. The authors systematically investigated the clinical phenotype of congenital motor nystagmus in a Chinese population. The study provides further evidence for clinical diagnosis and differential diagnosis and genetic counseling.
本研究的目的是鉴定 FRMD7(FERM 结构域包含 7)基因中的新突变,并对中国先天性眼球震颤患者的临床特征进行分析。为此,从中山大学中山眼科中心儿科和遗传科眼遗传病数据库中选择了 18 例先天性眼球震颤患者。采用直接测序法分析 FRMD7 基因的外显子和相邻内含子。采用异源双链-单链构象多态性分析 96 名无关正常对照和基因筛查阳性患者。采用裂隙灯照相术、眼底照相术、光学相干断层扫描和视网膜电图对先天性眼球震颤的临床特征进行分析。作者注意到,在 18 例先天性眼球震颤患者中,有 7 种 FRMD7 基因突变(6 种新突变)。筛查率为 38.89%,包括 c.41_43delAGA(p.13-15delK);c.473T>A(p.I158N);c.605T>A(p.I202N);c.580G>T(p.A194S);c.811T>A(p.C271S);c.1493insA(p.Y498X);c.57+1G>A(切片突变)。96 名正常对照中均未发现这些突变。这些结果丰富了 FRMD7 的基因突变谱。作者系统地研究了中国人群先天性眼球震颤的临床表型。该研究为临床诊断和鉴别诊断及遗传咨询提供了进一步的证据。