遗传性粘液上皮发育不良眼部受累的长期随访。

IF 1.2 4区 医学 Q3 OPHTHALMOLOGY Journal of Aapos Pub Date : 2024-09-13 DOI:10.1016/j.jaapos.2024.103997
Sepehr Feizi, Mohammadreza Tahavvori, Seyed-Bagher Hosseini, Goldis Espandar, Peyman Mohammadi Torbati, Hamed Esfandiari
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引用次数: 0

摘要

一名11个月大的非瘢痕性脱发男孩因4个月大时畏光而被转诊至眼科进行评估。全基因组测序发现,SREBF1基因存在杂合突变,确诊为遗传性粘液上皮发育不良。眼部检查发现睑板腺功能障碍、角膜表层血管扩张和混浊。角膜巩膜缘的印迹细胞学检查发现了非典型上皮细胞。患者接受了睑板腺功能障碍、干眼症和眼表炎症的治疗。经过适当的治疗和 7 年的密切随访,角膜混浊得到了很大改善。
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Long-term follow-up of ocular involvement in hereditary mucoepithelial dysplasia.

An 11-month-old boy with nonscarring alopecia was referred for ophthalmic evaluation because of photophobia from the age of 4 months. Whole-exome sequencing identified a heterozygous mutation in the SREBF1 gene, confirming the diagnosis of hereditary mucoepithelial dysplasia. Ocular examination revealed meibomian gland dysfunction and superficial corneal vascularization and opacity. Impression cytology of the sclerocorneal limbus revealed atypical epithelial cells. The patient received treatment for meibomian gland dysfunction, dry eye, and ocular surface inflammation. With appropriate management and close follow-up over 7 years, corneal opacity improved greatly.

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来源期刊
Journal of Aapos
Journal of Aapos 医学-小儿科
CiteScore
2.40
自引率
12.50%
发文量
159
审稿时长
55 days
期刊介绍: Journal of AAPOS presents expert information on children''s eye diseases and on strabismus as it affects all age groups. Major articles by leading experts in the field cover clinical and investigative studies, treatments, case reports, surgical techniques, descriptions of instrumentation, current concept reviews, and new diagnostic techniques. The Journal is the official publication of the American Association for Pediatric Ophthalmology and Strabismus.
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