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利妥昔单抗成功治疗1例合并甲状腺功能障碍性眼病的僵人综合征患者。

Successful treatment with rituximab in a patient with stiff-person syndrome complicated by dysthyroid ophthalmopathy.

作者信息

Katoh Nagaaki, Matsuda Masayuki, Ishii Wataru, Morita Hiroshi, Ikeda Shu-ichi

机构信息

Department of Medicine (Neurology and Rheumatology), Shinshu University School of Medicine, Matsumoto.

出版信息

Intern Med. 2010;49(3):237-41. doi: 10.2169/internalmedicine.49.2821. Epub 2010 Feb 1.

Abstract

We report a patient with stiff-person syndrome and insulin-dependent diabetes mellitus with anti-glutamic acid decarboxylase (GAD) antibody, who suddenly complained of diplopia due to dysthyroid ophthalmopathy. Therapeutic efficacy of plasmapheresis and high-dose intravenous immunoglobulin was transient. After starting administration of rituximab, the patient showed obvious improvement of muscle spasms due to stiff-person syndrome and ophthalmoplegia following quick depletion of CD20-positive cells in peripheral blood. The anti-GAD and anti-thyroid antibodies decreased slowly. This drug might be a potent therapeutic option for refractory patients with stiff-person syndrome, particularly in those associated with dysthyroid ophthalmopathy.

摘要

我们报告了一名患有僵人综合征和胰岛素依赖型糖尿病且伴有抗谷氨酸脱羧酶(GAD)抗体的患者,该患者因甲状腺功能障碍性眼病突然出现复视。血浆置换和大剂量静脉注射免疫球蛋白的治疗效果是短暂的。开始使用利妥昔单抗后,随着外周血中CD20阳性细胞迅速减少,患者僵人综合征所致的肌肉痉挛和眼肌麻痹明显改善。抗GAD抗体和抗甲状腺抗体缓慢下降。这种药物可能是难治性僵人综合征患者的一种有效治疗选择,尤其是那些伴有甲状腺功能障碍性眼病的患者。

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