Suppr超能文献

由d178n突变引起的致命性家族性失眠病例,其表型与桥本脑病相似。

Case of fatal familial insomnia caused by a d178n mutation with phenotypic similarity to Hashimoto's encephalopathy.

作者信息

Stevens Jessica M, Levine Matthew R, Constantino Anne E, Motamedi Gholam K

机构信息

Department of Neurology, MedStar Georgetown University Hospital, Washington, District of Columbia, USA.

出版信息

BMJ Case Rep. 2018 Jul 15;2018:bcr-2018-225155. doi: 10.1136/bcr-2018-225155.

Abstract

Fatal familial insomnia (FFI) is a rare prion disease commonly inherited in an autosomal dominant pattern from a mutation in the PRioN Protein (PRNP) gene. Hashimoto's encephalopathy (HE) is characterised by encephalopathy associated with antithyroid peroxidase (TPO) or antithyroglobulin (Tg) antibodies. These two conditions characteristically have differing clinical presentations with dramatically different clinical course and outcomes. Here, we present a case of FFI mimicking HE. A woman in her 50s presented with worsening confusion, hallucinations, tremor and leg jerks. Several maternal relatives had been diagnosed with FFI, but the patient had had negative genetic testing for PRNP. MRI of brain, cervical and thoracic spine were unremarkable except for evidence of prior cervical transverse myelitis. Cerebrospinal fluid analysis was normal. Anti-TPO and anti-Tg antibodies were elevated. She was started on steroids for possible HE and showed improvement in symptoms. Following discharge, the results of her PRNP gene test returned positive for variant p.Asp178Asn.

摘要

致死性家族性失眠症(FFI)是一种罕见的朊病毒病,通常以常染色体显性模式遗传,由朊病毒蛋白(PRNP)基因突变引起。桥本脑病(HE)的特征是与抗甲状腺过氧化物酶(TPO)或抗甲状腺球蛋白(Tg)抗体相关的脑病。这两种病症的典型临床表现不同,临床病程和结果也有显著差异。在此,我们报告一例疑似HE的FFI病例。一名50多岁的女性出现意识混乱、幻觉、震颤和腿部抽搐加重的症状。她的几位母系亲属被诊断患有FFI,但该患者的PRNP基因检测结果为阴性。除了既往有颈髓横贯性脊髓炎的证据外,脑部、颈椎和胸椎的MRI检查均无异常。脑脊液分析正常。抗TPO和抗Tg抗体升高。她开始接受类固醇治疗以治疗可能的HE,症状有所改善。出院后,她的PRNP基因检测结果显示p.Asp178Asn变异呈阳性。

相似文献

3
Fatal insomnia: the elusive prion disease.致死性失眠症:难以捉摸的朊病毒病。
BMJ Case Rep. 2021 Jun 22;14(6):e241289. doi: 10.1136/bcr-2020-241289.
9
Familial and sporadic fatal insomnia.家族性和散发性致命性失眠症。
Lancet Neurol. 2003 Mar;2(3):167-76. doi: 10.1016/s1474-4422(03)00323-5.

本文引用的文献

3
A clinical approach to diagnosis of autoimmune encephalitis.自身免疫性脑炎的临床诊断方法
Lancet Neurol. 2016 Apr;15(4):391-404. doi: 10.1016/S1474-4422(15)00401-9. Epub 2016 Feb 20.
5
A proposal of new diagnostic pathway for fatal familial insomnia.致命家族性失眠症新诊断途径的提出。
J Neurol Neurosurg Psychiatry. 2014 Jun;85(6):654-9. doi: 10.1136/jnnp-2013-305978. Epub 2013 Nov 18.
7
The genetics of prion diseases.朊病毒疾病的遗传学。
Genet Med. 2010 Apr;12(4):187-95. doi: 10.1097/GIM.0b013e3181cd7374.
9
Prominent corticosteroid disturbance in experimental prion disease.
Eur J Neurosci. 2006 May;23(10):2723-30. doi: 10.1111/j.1460-9568.2006.04801.x.

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