A Rare Presentation of Bilateral Vogt-Koyanagi-Harada (VKH) Disease in a Patient with Unilateral Limbal Stem Cell Deficiency (LSCD) with Corneal Perforation

S. Kodavoor, S. Balajee, R. Dandapani, M. Rajamani, Thenarasun Seethalakshmi Asaithambi
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Abstract

Purpose-To report a rare presentation of Vogt-Koyanagi-Harada (VKH) disease in a 55 year old female patient with unilateral limbal stem cell deficiency (LSCD) with corneal perforation. Observations-Patient presented to the out patient department with recurrent episodes of watering, pain, redness in the left eye. Examination revealed LSCD with areas of pannus and infiltration. She was treated medically. Patient came back after a year with a corneal perforation in her left eye. The perforation was sealed using cyanoacrylate glue. Six weeks later, she presented with a drop in vision in both eyes. Optical Coherence Tomography (OCT) in both eyes showed multiple sub retinal fluid pockets and Fundus Fluorescein Angiography (FFA) revealed multiple pinpoint leakages typical of Harada disease. The patient was investigated for systemic associations, which were found to be normal. She was managed with tapering doses of oral steroids. OCT showed resolution of fluid pockets at one month. There was a good control of the disease, with no signs of recurrence at 6 months follow up. Conclusion-The clinical association between the two entities has not been published in literature till date. Further data or reports on such cases would help throw light on any possible association between LSCD and VKH disease.
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单侧角膜缘干细胞缺乏症(LSCD)合并角膜穿孔患者罕见的双侧Vogt-Koyanagi-Harada (VKH)病
目的:报告一例55岁女性单侧角膜缘干细胞缺乏症(LSCD)合并角膜穿孔的罕见Vogt-Koyanagi-Harada (VKH)病。观察:患者就诊时左眼反复流泪、疼痛、发红。检查显示LSCD伴脓包和浸润。她接受了治疗。病人一年后因左眼角膜穿孔回来。穿孔处用氰基丙烯酸酯胶密封。六周后,她的双眼视力下降。双眼光学相干断层扫描(OCT)显示多个视网膜下液袋,眼底荧光素血管造影(FFA)显示多处原田病典型的针状渗漏。检查患者的全身关联,发现正常。她口服类固醇的剂量逐渐减少。OCT显示1个月后液体袋消失。病情控制良好,随访6个月无复发迹象。结论:两种实体之间的临床关联尚未在文献中发表。有关此类病例的进一步数据或报告将有助于阐明LSCD与VKH疾病之间的任何可能关联。
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