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难治性全身型重症肌无力一例的临床经过及治疗:胸腺切除术的疗效。

The Clinical Course and Treatment of a Case of Refractory Systemic Juvenile Myasthenia Gravis Successfully Treated with Thymectomy.

机构信息

Department of Pediatric Neurology, Miyagi Children's Hospital.

Department of Pediatrics, Tokyo Women's Medical University School of Medicine.

出版信息

Tohoku J Exp Med. 2024 Jan 23;262(1):29-31. doi: 10.1620/tjem.2023.J094. Epub 2023 Nov 16.

Abstract

Juvenile myasthenia gravis (JMG) exhibits a more favorable response to glucocorticoids and has a better prognosis than adult myasthenia gravis. However, no established treatment exists for refractory JMG. Although thymectomy has been performed in several patients with refractory systemic JMG, there are few detailed clinical descriptions of patients who underwent thymectomy. Here, we present the case of a 10-year-old boy with refractory systemic JMG who was successfully treated with thymectomy. The patient developed symptoms, including dysphagia, malaise, diurnal ptosis, and weakness in the trunk muscles, and he was diagnosed with generalized JMG. Despite undergoing various treatments, including steroids, tacrolimus, steroid pulse therapy, intravenous immunoglobulin, azathioprine (AZT), and rituximab, his symptoms did not improve. Therefore, he underwent a thoracoscopic thymectomy 24 months after disease onset. Thymectomy led to remission, as demonstrated by a significant reduction in the quantitative myasthenia gravis score and anti-acetylcholine receptor antibody levels, which persisted for 43 months after surgery. Our case demonstrates the effectiveness of thymectomy in systemic JMG patients with positive anti-acetylcholine receptor antibodies, despite therapeutic failure with AZT and rituximab, within 2 years of disease onset.

摘要

青少年重症肌无力(JMG)对糖皮质激素的反应更为有利,预后优于成人重症肌无力。然而,目前对于难治性 JMG 尚无既定的治疗方法。尽管胸腺切除术已在几例难治性系统性 JMG 患者中进行,但对于接受胸腺切除术的患者的详细临床描述很少。在这里,我们报告了一例 10 岁男孩难治性系统性 JMG 成功接受胸腺切除术治疗的病例。该患者出现吞咽困难、乏力、日间上睑下垂和躯干肌无力等症状,被诊断为全身型 JMG。尽管接受了各种治疗,包括类固醇、他克莫司、类固醇脉冲治疗、静脉注射免疫球蛋白、硫唑嘌呤(AZT)和利妥昔单抗,但症状并未改善。因此,他在发病后 24 个月接受了胸腔镜胸腺切除术。胸腺切除术导致了缓解,定量重症肌无力评分和抗乙酰胆碱受体抗体水平显著降低,并且在手术后 43 个月持续存在。我们的病例表明,在发病后 2 年内,即使 AZT 和利妥昔单抗治疗失败,对于抗乙酰胆碱受体抗体阳性的系统性 JMG 患者,胸腺切除术仍然有效。

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