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父子相继发生 Vogt-Koyanagi-Harada 综合征。

Successive onset of Vogt-Koyanagi-Harada syndrome in father and son.

机构信息

Tangshan Institute of Ophthalmology, Tangshan Eye Hospital, 063000, Hebei, China.

North China University of Science and Technology Affiliated Eye Hospital, 063000, Hebei, China.

出版信息

BMC Ophthalmol. 2023 Mar 21;23(1):113. doi: 10.1186/s12886-023-02851-2.

Abstract

BACKGROUND

Vogt‒Koyanagi‒Harada (VKH) disease is a multifactorial systemic autoimmune disorder against melanocytes that is characterized by panuveitis. Familial occurrence of VKH disease is rare. Here, we report two cases of a father and his son with characteristic manifestations of VKH disease.

CASE PRESENTATION

A 53-year-old male with typical clinical symptoms of VKH disease was referred to Tangshan Eye Hospital. Examination showed the presence of ciliochoroidal effusion and exudative retinal detachment in both eyes. The patient was given intravenous methylprednisolone 120 mg for 2 days and intravenous methylprednisolone 80 mg for 1 day followed by 48 mg (1 mg/kg/day) oral methylprednisolone daily, accompanied by oral azathioprine 50 mg daily. Cycloplegic agent (0.5% tropicamide three times daily [TID]) was added. The patient was free of symptoms and recurrence within more than 1-year-follow-up period, the best corrected visual acuity (BVCA) was increased and maintained in both eyes with complete resolution of subretinal fluid. One year and nine months later, case 2 (his son) also presented with the typical clinical symptoms of VKH disease at 29 years of age. The son also recovered from VKH disease after routine and standard treatment.

CONCLUSIONS

To the best of our knowledge, this is the first VKH disease case report of a father-son relationship. Although genetic factors have been demonstrated to be involved in the pathogenesis of VKH disease, the different inheritance modes of VKH patients need to be further explored and studied.

摘要

背景

Vogt-小柳-原田(Vogt-Koyanagi-Harada,VKH)病是一种多因素的全身自身免疫性疾病,针对黑色素细胞,其特征是全葡萄膜炎。家族性 VKH 病很少见。在这里,我们报告了两例具有 VKH 病典型表现的父子病例。

病例介绍

一名 53 岁男性,具有 VKH 病的典型临床症状,被转诊至唐山眼科医院。检查显示双眼睫状体脉络膜渗出和渗出性视网膜脱离。患者接受了 2 天 120mg 静脉注射甲基强的松龙和 1 天 80mg 静脉注射甲基强的松龙,随后每天口服 48mg(1mg/kg/天)甲基强的松龙,并每天口服 50mg 硫唑嘌呤。添加了睫状肌麻痹剂(0.5%托吡卡胺,每日 3 次[TID])。患者在超过 1 年的随访期内无症状和复发,最佳矫正视力(BVCA)在双眼增加并维持,视网膜下液完全消退。1 年零 9 个月后,病例 2(他的儿子)也在 29 岁时出现了 VKH 病的典型临床症状。儿子在常规和标准治疗后也从 VKH 病中康复。

结论

据我们所知,这是首例父子关系的 VKH 病病例报告。尽管遗传因素已被证明参与了 VKH 病的发病机制,但 VKH 患者的不同遗传模式仍需要进一步探索和研究。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6eae/10032000/4eed157c3796/12886_2023_2851_Fig1_HTML.jpg

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