Al-Moujahed Ahmad, Hien Doan Luong, Akhavanrezayat Amir, Pham Brandon Huy, Tuong Ngoc Than Trong, Doan Huy Luong, Yaşar Çigdem, Lajevardi Sherin, Nguyen Huy Vu, Nguyen Quan Dong
Byers Eye Institute, Stanford University, Palo Alto, CA, USA.
Pham Ngoc Thach University of Medicine, Saigon, Viet Nam.
Am J Ophthalmol Case Rep. 2021 Feb 5;21:101027. doi: 10.1016/j.ajoc.2021.101027. eCollection 2021 Mar.
To describe a case of ischemic retinal vasculitis in Adamantiades-Behçet disease (ABD) that demonstrated significant resolution of retinal ischemia following treatment with nicotinic acid and infliximab.: A 12-year-old male with a history of recurrent oral ulcers, fevers, and failure to thrive was admitted to the hospital with fever, oral and perirectal mucositis, and poor oral intake one month before presentation to uveitis clinic. He was suspected to have ABD and was treated with three doses of intravenous (IV) methylprednisolone (30 mg/kg/day) which led to improvement in his systemic symptoms. One week after admission, he complained of decreased vision in both eyes (OU), during which he was found to have anterior uveitis in OU and was referred to the Uveitis Clinic. Upon examination, his visual acuity was 20/80 in OU. Intraocular pressures were within normal limits. Anterior chamber evaluation revealed 0.5+ cells and 1.5+ flare in OU. Posterior examination revealed pale optic nerve, sclerosis and vascular sheathing of retinal arteries, and collateral vessels in OU. Fluorescein angiography (FA) showed optic disc leakage and widespread retinal ischemia in OU. The patient was diagnosed with retinal occlusive vasculitis associated with ABD. He was initially treated with infliximab (5 mg/kg), systemic methylprednisolone, and mycophenolate mofetil. Three months later, his BCVA improved to 20/70 OU with slight improvement of retinal ischemia on FA. Nicotinic acid was added to his treatment regimen. Due to logistic challenges, he did not receive infliximab treatment during the subsequent three months. However, three months after beginning nicotinic acid therapy, FA revealed significant improvement of his retinal ischemia OU. To our knowledge, the index report is the first to show that nicotinic acid may improve retinal ischemia in vaso-occlusive retinal vasculitis and be an integral part of the treatment regimen of this sight-threatening condition.
描述一例白塞病(ABD)合并缺血性视网膜血管炎的病例,该病例显示在接受烟酸和英夫利昔单抗治疗后视网膜缺血得到显著缓解。:一名12岁男性,有复发性口腔溃疡、发热及生长发育迟缓病史,在前往葡萄膜炎诊所就诊前1个月,因发热、口腔及直肠周围粘膜炎、口腔摄入不良入院。他被怀疑患有ABD,并接受了3剂静脉注射甲基强的松龙(30mg/kg/天)治疗,全身症状有所改善。入院1周后,他主诉双眼视力下降,检查发现双眼前部葡萄膜炎,遂转诊至葡萄膜炎诊所。检查时,他双眼视力为20/80。眼压在正常范围内。前房检查显示双眼有0.5+细胞和1.5+闪光。后部检查显示双眼视神经苍白、视网膜动脉硬化和血管鞘形成以及侧支血管。荧光素血管造影(FA)显示双眼视盘渗漏和广泛的视网膜缺血。该患者被诊断为与ABD相关的视网膜闭塞性血管炎。他最初接受英夫利昔单抗(5mg/kg)、全身用甲基强的松龙和霉酚酸酯治疗。3个月后,他的最佳矫正视力(BCVA)提高到双眼20/70,FA显示视网膜缺血略有改善。在他的治疗方案中加入了烟酸。由于后勤方面的困难,在随后的3个月里他没有接受英夫利昔单抗治疗。然而,在开始烟酸治疗3个月后,FA显示他双眼的视网膜缺血有显著改善。据我们所知,本报告首次表明烟酸可能改善血管闭塞性视网膜血管炎中的视网膜缺血,并且是这种威胁视力疾病治疗方案的一个组成部分。