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散发型晚发性杆状体肌病伴意义未明的单克隆丙种球蛋白血症 1 例:自体造血干细胞移植后神经症状的长期观察。

A case of sporadic late-onset nemaline myopathy with monoclonal gammopathy of undetermined significance: long-term observation of neurological symptoms after autologous stem-cell transplantation.

机构信息

Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan.

Department of Hematology, Japanese Red Cross Nagoya Daiichi Hospital, Nagoya, Japan.

出版信息

Nagoya J Med Sci. 2021 Aug;83(3):641-647. doi: 10.18999/nagjms.83.3.641.

DOI:10.18999/nagjms.83.3.641
PMID:34552296
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8438005/
Abstract

A 47-year-old woman presented with progressive limb weakness. A neurological examination revealed proximal dominant symmetrical muscle weakness in her limbs, and electromyography revealed complex repetitive discharges and short motor unit potentials with positive sharp waves in the biceps. We observed early recruitment in the quadriceps, and laboratory tests revealed normal creatine kinase. Serum protein electrophoresis showed monoclonal IgG-lambda, but the bone marrow aspiration specimen was normal. A muscle biopsy revealed nemaline rod accumulations in the muscle fibers; based on the results, we diagnosed the patient with sporadic late-onset nemaline myopathy with monoclonal gammopathy of undetermined significance (SLONM-MGUS). We administered repeated intravenous immunoglobulin, but her limb weakness continued, and she developed a restrictive ventilatory defect. The patient received melphalan, followed by autologous stem-cell transplantation (ASCT). Her upper extremity strength and respiratory capability improved within one year after ASCT; however, it was not until six years after ASCT that her atrophied lower extremities strengthened. A discrepancy in the timeline of treatment response between the upper or respiratory muscles and the atrophied lower limb was characteristic in the patient, suggesting that the efficacy of ASCT on SLONM-MGUS should be evaluated in the long term, especially in severely atrophied muscles. In addition, this case showed that ASCT for SLOMN-MGUS is an effective treatment option in Asian populations.

摘要

一位 47 岁女性因进行性四肢无力就诊。神经系统检查显示四肢近端为主的对称性肌肉无力,肌电图显示二头肌中复杂重复放电和短运动单位电位伴有正锐波。我们观察到股四头肌早期募集,实验室检查发现肌酸激酶正常。血清蛋白电泳显示单克隆 IgG-λ,但骨髓抽吸标本正常。肌肉活检显示肌纤维中有杆状体蓄积;根据结果,我们诊断患者为散发性晚发性杆状体肌病伴意义未明单克隆丙种球蛋白血症(SLONM-MGUS)。我们给予重复静脉注射免疫球蛋白,但她的四肢无力持续存在,并出现限制性通气缺陷。患者接受了美法仑,随后进行了自体干细胞移植(ASCT)。ASCT 后 1 年内,她的上肢力量和呼吸能力得到改善;然而,直到 ASCT 后 6 年,她萎缩的下肢才开始恢复力量。该患者的上肢或呼吸肌与萎缩的下肢之间的治疗反应时间存在差异,这表明 ASCT 对 SLONM-MGUS 的疗效应在长期内进行评估,尤其是在严重萎缩的肌肉中。此外,该病例表明 ASCT 治疗 SLONM-MGUS 是亚洲人群的一种有效治疗选择。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/60e7/8438005/ad830229e6bf/2186-3326-83-0641-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/60e7/8438005/ad830229e6bf/2186-3326-83-0641-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/60e7/8438005/ad830229e6bf/2186-3326-83-0641-g001.jpg

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本文引用的文献

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Neurology. 2019 Jul 16;93(3):e298-e305. doi: 10.1212/WNL.0000000000007777. Epub 2019 Jun 5.
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Monoclonal gammopathy with both nemaline myopathy and amyloid myopathy.伴有杆状体肌病和淀粉样变肌病的单克隆丙种球蛋白病
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Sporadic late-onset nemaline myopathy: clinico-pathological characteristics and review of 76 cases.
散发性迟发性杆状体肌病:76例临床病理特征及文献复习
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[Two cases of sporadic late onset nemaline myopathy effectively treated with immunotherapy].[两例散发性晚发型杆状体肌病经免疫治疗有效]
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