Vishnevskia-Dai Vicktoria, Chapman Joav, Sheinfeld Roee, Sharon Tal, Huna-Baron Ruth, Manor Riri S, Shoenfeld Yehuda, Zloto Ofira
Goldschleger Eye Institute Department of Neurology, Sagol Neuroscience Center, Tel Hashomer Department of Ophthalmology, Sheba Medical Center, Tel Hshomer Zabludowicz Center for Autoimmune Diseases, Sheba Medical Center, Tel Hashomer, are affiliated to the Sackler faculty of medicine Tel Aviv University, Israel.
Medicine (Baltimore). 2016 Oct;95(43):e5223. doi: 10.1097/MD.0000000000005223.
Susac syndrome is a rare condition characterized by the clinical triad of central nervous system (CNS) dysfunction, sensorineural hearing impairment, and branch retinal artery occlusion (BRAO). The purpose of this study is to examine the demographics, clinical characteristics, treatment, and long-term prognosis of Susac syndrome. The data recorded for all Susac syndrome patients treated at the Sheba Medical Center between 1998 and 2014 included demographics, clinical signs at presentation and during the disease course, imaging findings, treatment, and prognosis.Susac syndrome was diagnosed in 10 patients (age range 30-45 years). Only 2 patients presented with the full triad and 7 patients developed the full triad during mean follow-up period of 35 months. The average time to full triad was 7 months. Based on our observations at presentation, we divided the disease course into suspected, incomplete, and complete Susac syndrome. All 10 patients were treated at diagnosis with a pulse of high-dose intravenous methylprednisolone. There was improvement in visual acuity and visual field at the end of follow-up compared to baseline, but it was not statistically significant (P = 0.479 and P = 0.053, respectively). Five patients remained with neurological damage, and 5 patients had no improvement of their hearing loss at study closure. In conclusion, Susac syndrome is a rare condition that can mimic other disorders. The diagnosis is challenging because most patients do not initially present with the definitive triad. We suggest a clinical classification for the syndrome that may assist in early diagnosis.
Susac综合征是一种罕见疾病,其特征为中枢神经系统(CNS)功能障碍、感音神经性听力损害和视网膜分支动脉阻塞(BRAO)三联征。本研究的目的是探讨Susac综合征的人口统计学特征、临床特点、治疗方法及长期预后。记录了1998年至2014年在舍巴医疗中心接受治疗的所有Susac综合征患者的数据,包括人口统计学特征、就诊时及病程中的临床体征、影像学检查结果、治疗方法及预后情况。10例患者被诊断为Susac综合征(年龄范围30 - 45岁)。仅2例患者初诊时即表现出完整的三联征,7例患者在平均35个月的随访期内发展为完整的三联征。出现完整三联征的平均时间为7个月。根据我们在就诊时的观察,将病程分为疑似、不完全和完全性Susac综合征。所有10例患者在诊断时均接受了大剂量静脉注射甲泼尼龙冲击治疗。随访结束时与基线相比,视力和视野有所改善,但差异无统计学意义(分别为P = 0.479和P = 0.053)。5例患者仍存在神经损伤,5例患者在研究结束时听力损失无改善。总之,Susac综合征是一种罕见疾病,可模仿其他疾病。由于大多数患者初诊时未表现出明确的三联征,因此诊断具有挑战性。我们建议对该综合征进行临床分类,这可能有助于早期诊断。