Successful Treatment with Rituximab in a Patient with Stiff-Person Syndrome Complicated by Dysthyroid Ophthalmopathy
Successful Treatment with Rituximab in a Patient with Stiff-Person Syndrome Complicated by Dysthyroid Ophthalmopathy
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DOI：
10.2169/internalmedicine.49.2821
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发表时间：
2010-01-01
影响因子：
1.2
通讯作者：
Ikeda, Shu-ichi
中科院分区：
文献类型：
--
作者：
Katoh, Nagaaki;Matsuda, Masayuki;Ikeda, Shu-ichi
我们报告一位有抗谷氨酸脱羧酶（GAD）抗体的僵硬人症候群及胰岛素依赖型糖尿病患者，因甲状腺功能异常性眼病而突然主诉复视。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。在开始给予利妥昔单抗后，患者表现出由于僵硬人综合征和眼肌麻痹引起的肌肉痉挛的明显改善，随后外周血中的CD20阳性细胞快速耗竭。抗GAD和抗甲状腺抗体下降缓慢。这种药物可能是一个有效的治疗选择难治性患者僵硬的人综合征，特别是在那些与甲状腺功能障碍性眼病。
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.