Couriel D R, Beguelin G Z, Giralt S, De Lima M, Hosing C, Kharfan-Dabaja M A, Anagnostopoulos A, Champlin R
Department of Blood and Marrow Transplantation, University of Texas, MD Anderson Cancer Center, Houston, TX 77030, USA.
Bone Marrow Transplant. 2002 Oct;30(8):543-6. doi: 10.1038/sj.bmt.1703711.
Graft-versus-host disease (GVHD) remains a major complication of allogeneic hematopoietic stem cell transplantation. Polymyositis can occur in association with chronic GVHD and mimics the idiopathic form of the disease. We report two cases of chronic GVHD-associated polymyositis and review the published literature. The two patients presented 13 and 19 months after allogeneic transplantation with characteristic features of muscular hypotrophy, proximal muscle weakness, pain, elevated creatine phosphokinase (CPK), aldolase and SGPT. Interestingly, both patients had HLA DR52 genes, which is frequently reported in association with idiopathic polymyositis. Electromyogram (EMG) and muscle biopsy confirmed the diagnosis. Treatment with cyclosporine or tacrolimus resulted in complete and sustained remission of polymyositis in both cases. A review of the literature shows cyclosporine and steroids are well-described treatment options for patients with myositis in post transplant, as well as idiopathic cases. The duration of immunosuppressive treatment has varied in different reports, and there is a risk of recurrence when immunosuppression is tapered.
移植物抗宿主病(GVHD)仍然是异基因造血干细胞移植的主要并发症。多发性肌炎可与慢性GVHD相关联,并酷似特发性形式的该疾病。我们报告两例慢性GVHD相关的多发性肌炎病例并回顾已发表的文献。这两名患者在异基因移植后13个月和19个月出现,具有肌肉萎缩、近端肌无力、疼痛、肌酸磷酸激酶(CPK)、醛缩酶和谷丙转氨酶升高的特征性表现。有趣的是,两名患者均有HLA DR52基因,该基因在特发性多发性肌炎中常有报道。肌电图(EMG)和肌肉活检确诊了诊断。两例患者使用环孢素或他克莫司治疗后,多发性肌炎均完全且持续缓解。文献回顾显示,环孢素和类固醇是移植后肌炎患者以及特发性病例中已得到充分描述的治疗选择。不同报告中免疫抑制治疗的持续时间各不相同,且免疫抑制逐渐减量时有复发风险。