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异基因免疫性肌炎是严重慢性移植物抗宿主病后口腔鳞状细胞癌的副肿瘤并发症还是慢性移植物抗宿主病的一种表现?病例报告及文献讨论

Alloimmune Myositis as Paraneoplastic Complication of an Oral Squamous Cell Carcinoma After Severe Chronic Graft vs Host Disease or a Manifestation of Chronic Graft vs Host Disease? A Case Report and Literature Discussion.

作者信息

Woelfinger Pascal, Hauptrock Beate, Kriege Oliver, Wagner Wilfried, Heider Julia, Schwarting Andreas, Birklein Frank, Theobald Matthias, Wagner-Drouet Eva Maria

机构信息

Hematology, Oncology and Pneumology, UCT, University Medical Center Mainz, Mainz, Germany.

Hematology, Oncology and Pneumology, UCT, University Medical Center Mainz, Mainz, Germany.

出版信息

Transplant Proc. 2021 May;53(4):1365-1368. doi: 10.1016/j.transproceed.2020.11.010. Epub 2021 Jan 19.

DOI:10.1016/j.transproceed.2020.11.010
PMID:33478746
Abstract

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.

摘要

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。

相似文献

1
Alloimmune Myositis as Paraneoplastic Complication of an Oral Squamous Cell Carcinoma After Severe Chronic Graft vs Host Disease or a Manifestation of Chronic Graft vs Host Disease? A Case Report and Literature Discussion.异基因免疫性肌炎是严重慢性移植物抗宿主病后口腔鳞状细胞癌的副肿瘤并发症还是慢性移植物抗宿主病的一种表现?病例报告及文献讨论
Transplant Proc. 2021 May;53(4):1365-1368. doi: 10.1016/j.transproceed.2020.11.010. Epub 2021 Jan 19.
2
[Chronic graft-versus-host-disease involving the oral mucosa: clinical presentation and treatment].[涉及口腔黏膜的慢性移植物抗宿主病:临床表现与治疗]
Refuat Hapeh Vehashinayim (1993). 2008 Nov;25(4):19-27, 72.
3
[A case with myositis as a manifestation of chronic graft-versus-host-disease (GVHD) with severe muscle swelling developed after aggressive muscular exercise].[一例以肌炎为慢性移植物抗宿主病(GVHD)表现的病例,在剧烈肌肉运动后出现严重肌肉肿胀]
Rinsho Shinkeigaku. 2003 Mar;43(3):93-7.
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Early tapering of immunosuppressive agents after HLA-matched donor transplantation can improve the survival of patients with advanced acute myeloid leukemia.在人类白细胞抗原(HLA)匹配供体移植后早期减少免疫抑制剂用量可提高晚期急性髓系白血病患者的生存率。
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Allogeneic hematopoietic cell transplantation without myeloablative conditioning for patients with advanced hematologic malignancies.对晚期血液系统恶性肿瘤患者进行非清髓性预处理的异基因造血细胞移植。
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Successful treatment of meningeal graft-versus-host disease in a haematopoietic stem cell transplant recipient.造血干细胞移植受者脑膜移植物抗宿主病的成功治疗。
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引用本文的文献

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Oral squamous cell carcinoma arising from areas of Graft-versus-host disease: A systematic review.口腔黏膜鳞状细胞癌源于移植物抗宿主病:系统评价。
Med Oral Patol Oral Cir Bucal. 2024 Jan 1;29(1):e135-e144. doi: 10.4317/medoral.26133.