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一例Alport综合征患者的双侧前圆锥形晶状体:透明晶状体摘除成功后的临床与组织病理学相关性

Bilateral anterior lenticonus in a case of Alport syndrome: a clinical and histopathological correlation after successful clear lens extraction.

作者信息

Sonarkhan Shilpa, Ramappa Muralidhar, Chaurasia Sunita, Mulay Kaustubh

机构信息

Department of Pediatric Ophthalmology, L V Prasad Eye Institute, Hyderabad, Andhra Pradesh, India.

Department of Cornea and Anterior Segment Services, L V Prasad Eye Institute, Hyderabad, Andhra Pradesh, India.

出版信息

BMJ Case Rep. 2014 Jun 26;2014:bcr2013202036. doi: 10.1136/bcr-2013-202036.

Abstract

A 29-year-old woman presented with symptoms of gradual, progressive, painless diminution of vision in both eyes for past 8-10 years. On examination, uncorrected visual acuity was 20/60 in the right eye and 20/126 in the left eye. Anterior segment examination was essentially normal except for a conical protrusion in the pupillary axis of the anterior lens capsule with a clear underlying lens. During surgery, central protruded fragile lens capsule was meticulously handled to create well-centered continuous curvilinear capsulorhexis by using paediatric rhexis forceps. Histopathology of excised capsule showed markedly thinned lens capsule with normal epithelium. Electron microscopy of the anterior lens capsule showed multiple linear and irregular zones of dehiscence. Few of these had fibrillar, irregular electron-dense material and vacuoles; and adjacent cells were irregular suggestive of Alport syndrome. Postoperative course was uneventful and visual acuity improved to 20/20 unaided. Clear lens extraction is an effective means of rehabilitating visually symptomatic anterior lenticonus of Alport syndrome.

摘要

一名29岁女性,在过去8至10年中出现双眼视力逐渐、进行性、无痛性下降的症状。检查时,右眼未矫正视力为20/60,左眼为20/126。前段检查基本正常,除了前囊膜在瞳孔轴处有一个圆锥形突出,其下方晶状体清晰。手术过程中,使用小儿撕囊镊小心处理中央突出的脆弱晶状体囊膜,以制作中心良好的连续环形撕囊。切除囊膜的组织病理学显示晶状体囊膜明显变薄,上皮正常。前囊膜的电子显微镜检查显示有多个线性和不规则的裂开区域。其中少数有纤维状、不规则的电子致密物质和空泡;相邻细胞不规则,提示为奥尔波特综合征。术后过程顺利,视力提高到20/20,无需辅助。透明晶状体摘除是恢复奥尔波特综合征有视觉症状的前圆锥形晶状体的有效方法。

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