Suppr超能文献

苏萨克综合征(视网膜-耳蜗-脑微血管病):一名儿科患者的随访

Susac's Syndrome (Retinocochleocerebral Vasculopathy): Follow-up of a Pediatric Patient.

作者信息

Karalok Zeynep Selen, Taskin Birce Dilge, Guven Alev, Ucgul Cemile Atilgan, Aydin Omer Faruk

机构信息

Department of Pediatric Neurology, Ankara Children's Hematology Oncology Research and Training Hospital, Ankara, Turkey.

Department of Opthalmology, Ulucanlar Eyes Training and Research Hospital, Ankara, Turkey.

出版信息

J Pediatr Neurosci. 2017 Oct-Dec;12(4):374-377. doi: 10.4103/jpn.JPN_128_17.

Abstract

Susac's syndrome (SS) is a triad of encephalopathy, branch retinal artery occlusion (BRAO), and sensorineural hearing loss as a result of microvascular occlusions of the brain, retina, and inner ear. It is also a disorder of autoimmune endotheliopathy. SS usually affects young women between the age of 20 and 40 years. SS can be misdiagnosed as multiple sclerosis (MS) or acute disseminated encephalomyelitis (ADEM) because of similar findings. A 15-year-old girl presented in June 2015 with vomiting and severe headache. Cerebral magnetic resonance imaging revealed multiple lesions in the corpus callosum. Cerebrospinal fluid findings gave normal results. The initial diagnosis was MS and steroid (1000 mg/day) was given. She started to describe hallucinations and became paraplegic. She then underwent plasmapheresis five times without response. Her electroencephalogram was diffusely slow with 2-3 Hz delta rhythm at the frontal regions. Audiological examination showed that she had sensorineural hearing loss in her left ear. Ophthalmologic evaluation revealed BRAO in both eyes. On the basis of these findings, she was diagnosed with SS and treated with intravenous immunoglobulin (IVIG) and aspirin. After monthly treatment with IVIG for 6 months, the patient has almost fully recovered. SS should be kept in mind in the differential diagnosis of MS and ADEM.

摘要

Susac综合征(SS)是一种因脑、视网膜和内耳微血管闭塞而导致的脑病、视网膜分支动脉阻塞(BRAO)和感音神经性听力损失三联征。它也是一种自身免疫性内皮病变疾病。SS通常影响20至40岁的年轻女性。由于表现相似,SS可能被误诊为多发性硬化症(MS)或急性播散性脑脊髓炎(ADEM)。一名15岁女孩于2015年6月出现呕吐和严重头痛。脑磁共振成像显示胼胝体有多个病变。脑脊液检查结果正常。初步诊断为MS,并给予类固醇(1000毫克/天)治疗。她开始出现幻觉并发展为截瘫。随后她接受了5次血浆置换但无反应。她的脑电图弥漫性减慢,额叶区域有2至3赫兹的δ节律。听力检查显示她左耳有感音神经性听力损失。眼科评估显示双眼均有BRAO。基于这些发现,她被诊断为SS,并接受静脉注射免疫球蛋白(IVIG)和阿司匹林治疗。在每月接受IVIG治疗6个月后,患者几乎完全康复。在MS和ADEM的鉴别诊断中应考虑到SS。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8774/5890563/8358a323c55c/JPN-12-374-g001.jpg

相似文献

1
Susac's Syndrome (Retinocochleocerebral Vasculopathy): Follow-up of a Pediatric Patient.
J Pediatr Neurosci. 2017 Oct-Dec;12(4):374-377. doi: 10.4103/jpn.JPN_128_17.
3
Rituximab in the treatment of Susac's syndrome: Report of a case.
Eur J Ophthalmol. 2021 Sep;31(5):NP48-NP52. doi: 10.1177/1120672120924545. Epub 2020 May 7.
4
Recurrent Branch Retinal Artery Occlusions Revealing Susac's Syndrome.
Neuroophthalmology. 2021 May 3;46(1):50-53. doi: 10.1080/01658107.2021.1887286. eCollection 2022.
6
[Focus on Susac's syndrome. A pediatric case report].
J Fr Ophtalmol. 2014 Dec;37(10):804-11. doi: 10.1016/j.jfo.2014.09.005. Epub 2014 Nov 15.
8
Susac's syndrome diagnostic difficulties - the neurological point of view.
Neurol Neurochir Pol. 2022;56(2):141-147. doi: 10.5603/PJNNS.a2021.0082. Epub 2022 Jan 5.
9
Susac's syndrome: 1975-2005 microangiopathy/autoimmune endotheliopathy.
J Neurol Sci. 2007 Jun 15;257(1-2):270-2. doi: 10.1016/j.jns.2007.01.036. Epub 2007 Feb 28.
10
Susac's Syndrome--update.
J Neurol Sci. 2010 Dec 15;299(1-2):86-91. doi: 10.1016/j.jns.2010.08.032. Epub 2010 Sep 19.

引用本文的文献

1
Antithrombotic use in retinal artery occlusion: A narrative review.
Kaohsiung J Med Sci. 2025 Apr;41(4):e12938. doi: 10.1002/kjm2.12938. Epub 2025 Jan 16.
2
Susac Syndrome: A Rare cause of Bilateral Sensorineural hearing loss from North India-A Case Report with Review of literature.
Indian J Otolaryngol Head Neck Surg. 2022 Jun;74(2):196-204. doi: 10.1007/s12070-021-02853-w. Epub 2021 Sep 16.

本文引用的文献

1
Diagnosis and classification of Susac syndrome.
Autoimmun Rev. 2014 Apr-May;13(4-5):347-50. doi: 10.1016/j.autrev.2014.01.038. Epub 2014 Jan 11.
2
The primary vascular dysregulation syndrome: implications for eye diseases.
EPMA J. 2013 Jun 7;4(1):14. doi: 10.1186/1878-5085-4-14.
3
A case of isolated Susac occlusive retinal vasculitis.
J Neuroophthalmol. 2013 Sep;33(3):260-2. doi: 10.1097/WNO.0b013e31828bb64b.
5
Review of Susac syndrome.
Curr Opin Ophthalmol. 2011 Nov;22(6):472-6. doi: 10.1097/ICU.0b013e32834bbfeb.
6
Susac's syndrome: an update.
Autoimmun Rev. 2011 Jul;10(9):548-52. doi: 10.1016/j.autrev.2011.04.006. Epub 2011 Apr 15.
7
Branch retinal artery occlusion with visual field and multifocal erg in Susac syndrome: a case report.
Doc Ophthalmol. 2010 Dec;121(3):223-9. doi: 10.1007/s10633-010-9244-z. Epub 2010 Aug 26.
8
Microangiopathy of the brain and retina.
Neurology. 1979 Mar;29(3):313-6. doi: 10.1212/wnl.29.3.313.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验