Clinically amyopathic dermatomyositis manifested after the allogeneic haematopoietic stem cell transplantation: Case presentation and literature review
Clinically amyopathic dermatomyositis manifested after the allogeneic haematopoietic stem cell transplantation: Case presentation and literature review
复制标题
异基因造血干细胞移植后临床无肌病性皮肌炎的病例介绍及文献复习
DOI：
10.1093/mrcr/rxac060
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发表时间：
2022
影响因子：
0.8
通讯作者：
Masuzaki Hiroaki
中科院分区：
文献类型：
--
作者：
Miyagi Riko;Nakachi Sawako;Tamaki Yasutaro;Doi Mototsugu;Nakajima Tomo;Kitamura Sakiko;Tomori Shohei;Hanashiro Taeko;Tamaki Keita;Morichika Kazuho;Nishi Yukiko;Morishima Satoko;Fukushima Takuya;Masuzaki Hiroaki
临床上无肌病性皮肌炎（CADM）缺乏肌肉症状，与快速进展的间质性肺病相关。抗黑色素瘤分化相关基因5（MDA-5）抗体已被确定为疾病标记自身抗体。我们报告两例CADM表现后异基因造血干细胞移植（allo-HSCT）-例1：一个56岁的男性急性白血病接受allo-HSCT和例2：一个45岁的女性淋巴瘤患者接受allo-HSCT。由于移植后复发，她接受了供体淋巴细胞输注。在allo-HSCT或供体淋巴细胞输注后，出现Gottron丘疹，根据皮肤病学发现加上抗MDA-5抗体阳性，两名患者均被诊断为CADM，伴胸部计算机断层扫描显示与ILD一致的间质阴影。例2最初诊断为慢性移植物抗宿主病。他们的症状得到改善的免疫抑制剂与抗MDA-5抗体水平的伴随下降的组合。对于病例2，随后开始利妥昔单抗治疗淋巴瘤复发，导致抗MDA-5抗体水平大幅下降，皮疹和ILD改善。我们的病例提出了HSCT后出现CADM的可能性，强调了早期诊断的重要性，以避免注定进展为ILD。
Clinically amyopathic dermatomyositis (CADM) lacks muscle symptoms, associated with rapidly progressive interstitial lung disease. Anti-melanoma differentiation-associated gene 5 (MDA-5) antibody has been identified as a disease-labelling autoantibody. We report two cases of CADM manifested after the allogeneic haematopoietic stem cell transplantation (allo-HSCT)—Case 1: a 56-year-old man with acute leukaemia received the allo-HSCT and Case 2: a 45-year-old female patient with lymphoma received the allo-HSCT. She received donor lymphocyte infusion because of a post-transplant relapse. After allo-HSCT or donor lymphocyte infusion, Gottron papules emerged, and both patients were diagnosed as CADM based on dermatological findings coupled with the positivity of anti-MDA-5 antibody, accompanied by interstitial shadows consistent with ILD on chest computed tomography. Case 2 was initially diagnosed as a kind of chronic graft versus host disease. Their symptoms were improved by the combination of immunosuppressive agents with a concomitant decrease in anti-MDA-5 antibody levels. For Case 2, rituximab was subsequently started for relapse of lymphoma, resulting in a substantial decrease in the level of anti-MDA-5 antibody and improvement in rash and ILD. Our cases raise a possibility that CADM emerges after the HSCT, highlighting the importance of early diagnosis to avoid fated progression into ILD.