Woelfinger Pascal, Hauptrock Beate, Kriege Oliver, Wagner Wilfried, Heider Julia, Schwarting Andreas, Birklein Frank, Theobald Matthias, Wagner-Drouet Eva Maria
Hematology, Oncology and Pneumology, UCT, University Medical Center Mainz, Mainz, Germany.
Hematology, Oncology and Pneumology, UCT, University Medical Center Mainz, Mainz, Germany.
Transplant Proc. 2021 May;53(4):1365-1368. doi: 10.1016/j.transproceed.2020.11.010. Epub 2021 Jan 19.
A 53-year-old female patient with acute myeloid leukemia developed severe chronic graft vs host disease (cGVHD) of the oral mucosa after allogeneic hematopoietic stem cell transplantation with leukoplakia and relapsing oral squamous cell carcinoma (SCC) of the tongue. cGVHD needed long-lasting immunosuppressive therapy; SCC was treated with radiation and surgery. Acute myeloid leukemia remained in complete remission. The patient developed a myositis with pain of all muscles as well as paraparesis with elevated creatine kinase and C-reactive protein and detection of antiskeletal muscle autoantibodies 3500 days after hematopoietic stem cell transplantation. No other clinical features of chronic GVHD were apparent at this time. Symptoms disappeared after treatment with corticosteroids but relapsed while tapering. Weekly therapy with the B-cell-depleting antibody rituximab was started and administered for 6 weeks. Symptoms disappeared again but partly returned after some weeks, so therapy with azathioprine was started. During therapy with azathioprine slow tapering of corticosteroids was possible and clinical symptoms remained absent. Here we present a case report and review of the literature on alloimmune myositis as paraneoplastic complication of an oral SCC of the tongue after severe chronic GVHD or as late manifestation of chronic GVHD itself.
一名53岁的急性髓系白血病女性患者在异基因造血干细胞移植后发生了严重的口腔黏膜慢性移植物抗宿主病(cGVHD),伴有白斑和舌部复发性口腔鳞状细胞癌(SCC)。cGVHD需要长期的免疫抑制治疗;SCC采用放疗和手术治疗。急性髓系白血病仍处于完全缓解状态。该患者在造血干细胞移植3500天后出现了肌炎,所有肌肉疼痛,以及轻截瘫,肌酸激酶和C反应蛋白升高,并检测到抗骨骼肌自身抗体。此时没有其他慢性移植物抗宿主病的临床特征。使用皮质类固醇治疗后症状消失,但在逐渐减量时复发。开始每周使用耗竭B细胞的抗体利妥昔单抗治疗,并持续6周。症状再次消失,但几周后部分复发,因此开始使用硫唑嘌呤治疗。在使用硫唑嘌呤治疗期间,可以缓慢减少皮质类固醇的用量,临床症状也未再出现。在此,我们报告一例病例,并对文献进行综述,内容涉及异基因免疫性肌炎作为严重慢性移植物抗宿主病后舌部口腔鳞状细胞癌的副肿瘤并发症,或作为慢性移植物抗宿主病本身的晚期表现。