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僵肢综合征:一例报告

Stiff limb syndrome: a case report.

作者信息

Hajjioui Abderrazak, Benbouazza Karima, Faris Mohamed El Alaoui, Missaoui Amina, Hassouni Najia Hajjaj

机构信息

Physical Medicine and Rehabilitation, El Ayachi Hospital, Mohamed V University, Coast Road Street, Sale, 11000, Morocco.

出版信息

Cases J. 2010 Feb 12;3:60. doi: 10.1186/1757-1626-3-60.

DOI:10.1186/1757-1626-3-60
PMID:20205913
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC2829520/
Abstract

INTRODUCTION

Stiff limb syndrome is a clinical feature of the stiff person syndrome, which is a rare and disabling neurologic disorder characterized by muscle rigidity and episodic spasms that involve axial and limb musculature. It is an autoimmune disorder resulting in a malfunction of aminobutyric acid mediated inhibitory networks in the central nervous system. We describe a patient diagnosed by neurological symptoms of stiff limb syndrome with a good outcome after treatment, and a review of the related literature.

CASE PRESENTATION

A 49-year-old male patient presented with a progressive stiffness and painful spasms of his both legs resulting in a difficulty of standing up and walking. The diagnosis of stiff limb syndrome was supported by the dramatically positive response to treatment using diazepam 25 mg/day and baclofen 30 mg/day.

CONCLUSION

This clinical case highlights the importance of a therapeutic test to confirm the diagnosis of stiff limb syndrome especially when there is a high clinical suspicion with unremarkable electromyography.

摘要

引言

僵肢综合征是僵人综合征的一种临床特征,僵人综合征是一种罕见的致残性神经系统疾病,其特征为肌肉僵硬和发作性痉挛,累及躯干和四肢肌肉。它是一种自身免疫性疾病,导致中枢神经系统中γ-氨基丁酸介导的抑制网络功能失调。我们描述了一名经僵肢综合征神经症状诊断的患者,治疗后效果良好,并对相关文献进行了综述。

病例介绍

一名49岁男性患者出现双下肢进行性僵硬和疼痛性痉挛,导致站立和行走困难。使用地西泮25mg/天和巴氯芬30mg/天治疗后出现显著阳性反应,支持僵肢综合征的诊断。

结论

本临床病例强调了治疗性试验对确诊僵肢综合征的重要性,尤其是在临床高度怀疑但肌电图无明显异常的情况下。

相似文献

1
Stiff limb syndrome: a case report.僵肢综合征:一例报告
Cases J. 2010 Feb 12;3:60. doi: 10.1186/1757-1626-3-60.
2
Stiff Person Syndrome.僵人综合征
J Assoc Physicians India. 2015 Aug;63(8):81-2.
3
The stiff-person syndrome: an autoimmune disorder affecting neurotransmission of gamma-aminobutyric acid.僵人综合征:一种影响γ-氨基丁酸神经传递的自身免疫性疾病。
Ann Intern Med. 1999 Oct 5;131(7):522-30. doi: 10.7326/0003-4819-131-7-199910050-00008.
4
Review of 23 patients affected by the stiff man syndrome: clinical subdivision into stiff trunk (man) syndrome, stiff limb syndrome, and progressive encephalomyelitis with rigidity.对23例僵人综合征患者的回顾:临床分为僵躯干(人)综合征、僵肢综合征和进行性脑脊髓炎伴强直。
J Neurol Neurosurg Psychiatry. 1998 Nov;65(5):633-40. doi: 10.1136/jnnp.65.5.633.
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The stiff-person syndrome. Case report.僵人综合征。病例报告。
Minerva Anestesiol. 2002 Nov;68(11):861-5.
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A Rare Case of Childhood Stiff Person Syndrome Associated With Pleuropulmonary Blastoma.一例与肺胚细胞瘤相关的儿童僵人综合征罕见病例。
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A Stiff Person Syndrome Misdiagnosed as a Psychiatric Illness: A Case Report.一例被误诊为精神疾病的僵人综合征病例报告。
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Front Neurol. 2020 May 8;11:284. doi: 10.3389/fneur.2020.00284. eCollection 2020.
2
Stiff limb syndrome with lower limb myoclonus: A case report.伴有下肢肌阵挛的僵肢综合征:一例报告
Medicine (Baltimore). 2019 Dec;98(49):e18160. doi: 10.1097/MD.0000000000018160.

本文引用的文献

1
Progressive fluctuating muscular rigidity and spasm ("stiff-man" syndrome); report of a case and some observations in 13 other cases.进行性波动性肌肉僵硬和痉挛(“僵人”综合征);1例报告及对其他13例的一些观察
Proc Staff Meet Mayo Clin. 1956 Jul 25;31(15):421-7.
2
Stiff person syndrome and anesthesia: case report.僵人综合征与麻醉:病例报告
Anesth Analg. 2003 Aug;97(2):486-487. doi: 10.1213/01.ANE.0000072701.97699.79.
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The stiff-person syndrome. Case report.僵人综合征。病例报告。
Minerva Anestesiol. 2002 Nov;68(11):861-5.
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Stiff man syndrome and related conditions.僵人综合征及相关病症。
Mov Disord. 2002 Sep;17(5):853-66. doi: 10.1002/mds.10279.
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Stiff-man syndrome: a case report and review of the literature.僵人综合征:一例病例报告及文献综述
Psychosomatics. 2002 May-Jun;43(3):243-4. doi: 10.1176/appi.psy.43.3.243.
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High-dose intravenous immune globulin for stiff-person syndrome.大剂量静脉注射免疫球蛋白治疗僵人综合征。
N Engl J Med. 2001 Dec 27;345(26):1870-6. doi: 10.1056/NEJMoa01167.
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Stiff leg syndrome: case report.僵腿综合征:病例报告
Mov Disord. 2001 Nov;16(6):1189-93. doi: 10.1002/mds.1224.
8
The clinical spectrum of anti-GAD antibody-positive patients with stiff-person syndrome.抗谷氨酸脱羧酶(GAD)抗体阳性僵人综合征患者的临床谱
Neurology. 2000 Nov 28;55(10):1531-5. doi: 10.1212/wnl.55.10.1531.
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Plasmapheresis in the treatment of stiff-man syndrome.
N Engl J Med. 1989 Jun 1;320(22):1499. doi: 10.1056/nejm198906013202220.