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利妥昔单抗治疗僵人综合征。

Stiff-person syndrome treated with rituximab.

作者信息

Lobo Marcelo Evangelista, Araújo Marx Lincoln Barros, Tomaz Carlos Alberto Bezerra, Allam Nasser

机构信息

Department of Neurology, Hospital Universitário de Brasília, Brasilia, Brazil.

出版信息

BMJ Case Rep. 2010 Dec 13;2010:bcr0520103021. doi: 10.1136/bcr.05.2010.3021.

DOI:10.1136/bcr.05.2010.3021
PMID:22802263
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3029087/
Abstract

Stiff-person syndrome (SPS) is a rare neurological condition consisting of progressive and fluctuating rigidity of the axial muscles combined with painful spasms. The pathophysiology of SPS is not fully understood, but there seems to be an autoimmune component. The use of rituximab, a chimeric monoclonal antibody targeting CD20 protein in the surface of mature B cells, for the treatment of SPS is a recent therapeutical approach showing promising results. The authors present a case report of a 41-year-old female patient diagnosed with SPS who was treated with rituximab in a public hospital in Brasília, Brazil, showing a good and safe response to the treatment so far. Our data go along with some recent articles published in the literature.

摘要

僵人综合征(SPS)是一种罕见的神经系统疾病,表现为轴向肌肉进行性和波动性僵硬,并伴有疼痛性痉挛。SPS的病理生理学尚未完全明确,但似乎存在自身免疫成分。利妥昔单抗是一种靶向成熟B细胞表面CD20蛋白的嵌合单克隆抗体,将其用于治疗SPS是一种近期的治疗方法,已显示出有前景的结果。作者报告了一例41岁女性SPS患者的病例,该患者在巴西巴西利亚的一家公立医院接受了利妥昔单抗治疗,目前显示出对治疗良好且安全的反应。我们的数据与文献中最近发表的一些文章一致。

相似文献

1
Stiff-person syndrome treated with rituximab.利妥昔单抗治疗僵人综合征。
BMJ Case Rep. 2010 Dec 13;2010:bcr0520103021. doi: 10.1136/bcr.05.2010.3021.
2
Treatment of thyroid-associated orbitopathy with rituximab--a novel therapy for an old disease: case report and literature review.采用利妥昔单抗治疗甲状腺相关眼病——一种针对古老疾病的新型疗法:病例报告及文献复习。
Endocr Pract. 2010 Jul-Aug;16(4):677-85. doi: 10.4158/EP09385.RA.
3
Stiff person syndrome (SPS) complicated by respiratory failure: successful treatment with rituximab.僵人综合征(SPS)合并呼吸衰竭:利妥昔单抗治疗成功病例
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4
A Case of Stiff Person Syndrome: Immunomodulatory Effect of Benzodiazepines: Successful Rituximab and Tizanidine Therapy.僵人综合征一例:苯二氮䓬类药物的免疫调节作用:利妥昔单抗和替扎尼定治疗成功
Medicine (Baltimore). 2015 Jun;94(23):e954. doi: 10.1097/MD.0000000000000954.
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Rituximab improves not only back stiffness but also "stiff eyes" in stiff person syndrome: Implications for immune-mediated treatment.利妥昔单抗不仅能改善僵硬感,还能改善僵人综合征的“眼僵”:提示免疫介导的治疗作用。
J Neurol Sci. 2020 Jan 15;408:116506. doi: 10.1016/j.jns.2019.116506. Epub 2019 Nov 2.
6
Treatment of stiff person syndrome with rituximab.利妥昔单抗治疗僵人综合征
J Neurol Neurosurg Psychiatry. 2005 Jul;76(7):999-1001. doi: 10.1136/jnnp.2004.051144.
7
[Successful treatment with rituximab in a refractory Stiff-person syndrome].[利妥昔单抗成功治疗难治性僵人综合征]
Rev Neurol (Paris). 2012 Apr;168(4):375-8. doi: 10.1016/j.neurol.2011.09.008. Epub 2012 Feb 14.
8
Long-lived plasma cells and memory B cells produce pathogenic anti-GAD65 autoantibodies in Stiff Person Syndrome.在僵人综合征中,长寿浆细胞和记忆 B 细胞产生致病性抗谷氨酸脱羧酶 65 自身抗体。
PLoS One. 2010 May 26;5(5):e10838. doi: 10.1371/journal.pone.0010838.
9
Stiff-person syndrome: persistent elevation of glutamic acid decarboxylase antibodies despite successful treatment with rituximab.僵人综合征:尽管利妥昔单抗治疗成功,但谷氨酸脱羧酶抗体持续升高。
J Clin Rheumatol. 2010 Aug;16(5):237-9. doi: 10.1097/RHU.0b013e3181e931fa.
10
Rituximab treatment of stiff-person syndrome in a patient with thymoma, diabetes mellitus and autoimmune thyroiditis.利妥昔单抗治疗合并胸腺瘤、糖尿病和自身免疫性甲状腺炎的僵人综合征
J Clin Neurosci. 2010 Mar;17(3):389-91. doi: 10.1016/j.jocn.2009.06.015. Epub 2010 Jan 13.

引用本文的文献

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Rituximab in stiff-person syndrome with glutamic acid decarboxylase 65 autoantibody: a systematic review.利妥昔单抗治疗伴谷氨酸脱羧酶65自身抗体的僵人综合征:一项系统评价
J Neurol. 2025 May 24;272(6):417. doi: 10.1007/s00415-025-13157-2.
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An update on malignant tumor-related stiff person syndrome spectrum disorders: clinical mechanism, treatment, and outcomes.恶性肿瘤相关僵人综合征谱系障碍的最新进展:临床机制、治疗及预后
Front Neurol. 2023 Oct 4;14:1209302. doi: 10.3389/fneur.2023.1209302. eCollection 2023.
3
Paraneoplastic stiff-person syndrome with lung cancer: a case report and literature review.肺癌相关副肿瘤性僵人综合征:一例报告及文献复习
Am J Transl Res. 2023 Jan 15;15(1):330-335. eCollection 2023.
4
Stiff-Person Syndrome: A Treatment Update and New Directions.僵人综合征:治疗进展与新方向
Cureus. 2020 Dec 9;12(12):e11995. doi: 10.7759/cureus.11995.
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GAD antibodies in neurological disorders - insights and challenges.神经疾病中的 GAD 抗体:研究进展与挑战。
Nat Rev Neurol. 2020 Jul;16(7):353-365. doi: 10.1038/s41582-020-0359-x. Epub 2020 May 26.
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Stiff-person Syndrome with Waldenström Macroglobulinemia.僵人综合征合并华氏巨球蛋白血症
Intern Med. 2018 Nov 15;57(22):3313-3316. doi: 10.2169/internalmedicine.1043-18. Epub 2018 Jul 6.
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Evaluation of Treatment Outcomes in Patients with Stiff Person Syndrome with Rituximab vs. Standard of Care.利妥昔单抗与标准治疗对僵人综合征患者治疗效果的评估
Cureus. 2017 Jun 24;9(6):e1387. doi: 10.7759/cureus.1387.
8
Clinical Spectrum of Stiff Person Syndrome: A Review of Recent Reports.僵人综合征的临床谱:近期报告综述
Tremor Other Hyperkinet Mov (N Y). 2016 Mar 4;6:340. doi: 10.7916/D85M65GD. eCollection 2016.
9
Recent Advances and Review on Treatment of Stiff Person Syndrome in Adults and Pediatric Patients.成人及儿童僵人综合征治疗的最新进展与综述
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Anti-B-Cell Therapies in Autoimmune Neurological Diseases: Rationale and Efficacy Trials.自身免疫性神经疾病中的抗 B 细胞治疗:原理与疗效试验。
Neurotherapeutics. 2016 Jan;13(1):20-33. doi: 10.1007/s13311-015-0402-6.

本文引用的文献

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[Stiff-person syndrome: a case report].[僵人综合征:一例报告]
Rev Neurol. 2008;47(5):249-52.
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Treatment of stiff person syndrome with rituximab.利妥昔单抗治疗僵人综合征
J Neurol Neurosurg Psychiatry. 2005 Jul;76(7):999-1001. doi: 10.1136/jnnp.2004.051144.
3
Progressive encephalomyelitis with rigidity: a paraneoplastic presentation of oat cell carcinoma of the lung. Case report.进行性脑脊髓炎伴强直:肺癌燕麦细胞癌的副肿瘤表现。病例报告。
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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.
5
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.
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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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Botulinum toxin A improves muscle spasms and rigidity in stiff-person syndrome.A型肉毒杆菌毒素可改善僵人综合征中的肌肉痉挛和强直。
Mov Disord. 1997 Nov;12(6):1060-3. doi: 10.1002/mds.870120636.
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Treatment of spasticity with botulinum toxin: a double-blind study.肉毒杆菌毒素治疗痉挛:一项双盲研究。
Ann Neurol. 1990 Oct;28(4):512-5. doi: 10.1002/ana.410280407.