Liu X Y, Tao Y F, Mao Y K, Chen Z J, Wang Y, Hong Y F, Fan N
Xiamen Eye Center of Xiamen University, Fujian Provincial Key Laboratory of Ocular Surface & Corneal Diseases, Xiamen Municipal Key Laboratory of Ocular Surface & Corneal Diseases, Xiamen Municipal Key Laboratory of Ocular Diseases, Xiamen Clinical Research Center for Eye Diseases, Xiamen 361002, China.
Shenzhen Eye Hospital, the Second Clinical Medical College, Jinan University, Shenzhen 518000, China.
Zhonghua Yan Ke Za Zhi. 2024 Jan 11;60(1):78-83. doi: 10.3760/cma.j.cn112142-20231012-00134.
This case report presents a family with developmental glaucoma accompanied by microcornea resulting from novel mutations in the ADAMTS18 gene. The index case involves a 5-year-old twin brother, who, during a routine examination, exhibited elevated intraocular pressure persisting for over a month. The peak intraocular pressure reached approximately 25 mmHg (1 mmHg=0.133 kPa) in both eyes, with a corneal diameter of less than 10 mm. Ocular examination revealed an enlarged cup-to-disc ratio, and optical coherence tomography (OCT) demonstrated thinning of the retinal nerve fiber layer and ganglion cell layer. Ultrasound biomicroscopy combined with gonioscopy indicated partial angle closure and abnormal anterior chamber angle development. The ocular manifestations in the twin brother were consistent with those observed in the twin sister. The clinical diagnosis was bilateral developmental glaucoma with microcornea. Genetic sequencing identified two novel compound heterozygous mutations in the ADAMTS18 gene in the twins: Mutation 1 (M1) involving the variant site 1 (c.3436C>T:p.R1146W) and Mutation 2 (M2) involving the variant site 2 (c.1454T>G:p.F485C). Ocular examinations of four additional family members were normal. Genetic testing revealed that the twins' father and sister carried M1, while the index case's mother and brother carried M2. This report underscores a unique association between ADAMTS18 gene mutations and developmental glaucoma with microcornea within a familial context, emphasizing the importance of genetic screening for early diagnosis and targeted management strategies.
本病例报告介绍了一个患有发育性青光眼并伴有小角膜的家族,这是由ADAMTS18基因的新突变引起的。索引病例是一名5岁的双胞胎兄弟,在一次常规检查中,他的眼压持续升高超过一个月。双眼眼压峰值约为25 mmHg(1 mmHg = 0.133 kPa),角膜直径小于10 mm。眼部检查发现杯盘比增大,光学相干断层扫描(OCT)显示视网膜神经纤维层和神经节细胞层变薄。超声生物显微镜检查结合前房角镜检查提示部分房角关闭和前房角发育异常。双胞胎兄弟的眼部表现与双胞胎姐妹观察到的一致。临床诊断为双侧发育性青光眼合并小角膜。基因测序在双胞胎中发现了ADAMTS18基因的两个新的复合杂合突变:突变1(M1)涉及变异位点1(c.3436C>T:p.R1146W),突变2(M2)涉及变异位点2(c.1454T>G:p.F485C)。另外四名家庭成员的眼部检查正常。基因检测显示,双胞胎的父亲和姐姐携带M1,而索引病例的母亲和兄弟携带M2。本报告强调了ADAMTS18基因突变与家族性发育性青光眼合并小角膜之间的独特关联,强调了基因筛查对于早期诊断和靶向管理策略的重要性。