{"title":"多药免疫抑制治疗抗黑色素瘤分化相关基因5抗体阳性皮肌炎后的其他医源性免疫缺陷相关淋巴细胞增生性疾病1例报告","authors":"Shungo Mochizuki, Toshiki Nakajima, Shota Ohsumi, Aiko Ogura, Nozomi Akatsu, Noriyoshi Takebe, Kentaro Odani, Toshiyuki Kitano, Yoshitaka Imura","doi":"10.1093/mrcr/rxaf043","DOIUrl":null,"url":null,"abstract":"<p><p>Although other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPDs) are rare, they are important adverse effects of immunosuppressive therapies. Even though anti-melanoma differentiation association gene 5 (MDA5) antibody-positive dermatomyositis requires multidrug immunosuppressive therapy for interstitial pneumonia control, OIIA-LPD has rarely been reported. Moreover, central nervous system (CNS) OIIA-LPD has never been documented. Here, we report a case of CNS OIIA-LPD that may have been caused by treatment for MDA5 antibody-positive dermatomyositis. A 53-year-old woman was diagnosed with MDA5 dermatomyositis and treated for rapidly progressive interstitial lung disease using multidrug immunosuppressive therapy with prednisolone (PSL), tacrolimus, and intravenous cyclophosphamide pulse therapy. Seven months after treatment initiation, vomiting led to the discovery of a cerebellar tumour. The cerebellar tumour was histologically Epstein-Barr virus (EBV)-encoded small RNA-positive diffuse large B-cell lymphoma, with EBV-DNA being positive in the blood. The patient was diagnosed with OIIA-LPDs due to EBV reactivation. Chemotherapy, including high-dose methotrexate (MTX) and rituximab, prevented tumour recurrence without exacerbating interstitial lung disease. This is the first reported case of CNS OIIA-LPD with multidrug immunosuppression in a patient with MDA5 dermatomyositis. Chemotherapy, including high-dose MTX and rituximab, can be used for central OIIA-LPD without aggravating settled interstitial lung disease. The activity of MDA5 dermatomyositis during OIIA-LPD treatment may be managed with low-dose PSL.</p>","PeriodicalId":94146,"journal":{"name":"Modern rheumatology case reports","volume":" ","pages":""},"PeriodicalIF":0.9000,"publicationDate":"2025-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Other iatrogenic immunodeficiency-associated lymphoproliferative disorders after multidrug immunosuppressive therapy for anti-melanoma differentiation association gene 5 antibody-positive dermatomyositis: a case report.\",\"authors\":\"Shungo Mochizuki, Toshiki Nakajima, Shota Ohsumi, Aiko Ogura, Nozomi Akatsu, Noriyoshi Takebe, Kentaro Odani, Toshiyuki Kitano, Yoshitaka Imura\",\"doi\":\"10.1093/mrcr/rxaf043\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Although other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPDs) are rare, they are important adverse effects of immunosuppressive therapies. Even though anti-melanoma differentiation association gene 5 (MDA5) antibody-positive dermatomyositis requires multidrug immunosuppressive therapy for interstitial pneumonia control, OIIA-LPD has rarely been reported. Moreover, central nervous system (CNS) OIIA-LPD has never been documented. Here, we report a case of CNS OIIA-LPD that may have been caused by treatment for MDA5 antibody-positive dermatomyositis. A 53-year-old woman was diagnosed with MDA5 dermatomyositis and treated for rapidly progressive interstitial lung disease using multidrug immunosuppressive therapy with prednisolone (PSL), tacrolimus, and intravenous cyclophosphamide pulse therapy. Seven months after treatment initiation, vomiting led to the discovery of a cerebellar tumour. The cerebellar tumour was histologically Epstein-Barr virus (EBV)-encoded small RNA-positive diffuse large B-cell lymphoma, with EBV-DNA being positive in the blood. The patient was diagnosed with OIIA-LPDs due to EBV reactivation. Chemotherapy, including high-dose methotrexate (MTX) and rituximab, prevented tumour recurrence without exacerbating interstitial lung disease. This is the first reported case of CNS OIIA-LPD with multidrug immunosuppression in a patient with MDA5 dermatomyositis. Chemotherapy, including high-dose MTX and rituximab, can be used for central OIIA-LPD without aggravating settled interstitial lung disease. The activity of MDA5 dermatomyositis during OIIA-LPD treatment may be managed with low-dose PSL.</p>\",\"PeriodicalId\":94146,\"journal\":{\"name\":\"Modern rheumatology case reports\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":0.9000,\"publicationDate\":\"2025-07-25\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Modern rheumatology case reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1093/mrcr/rxaf043\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"RHEUMATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Modern rheumatology case reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1093/mrcr/rxaf043","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"RHEUMATOLOGY","Score":null,"Total":0}
Other iatrogenic immunodeficiency-associated lymphoproliferative disorders after multidrug immunosuppressive therapy for anti-melanoma differentiation association gene 5 antibody-positive dermatomyositis: a case report.
Although other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPDs) are rare, they are important adverse effects of immunosuppressive therapies. Even though anti-melanoma differentiation association gene 5 (MDA5) antibody-positive dermatomyositis requires multidrug immunosuppressive therapy for interstitial pneumonia control, OIIA-LPD has rarely been reported. Moreover, central nervous system (CNS) OIIA-LPD has never been documented. Here, we report a case of CNS OIIA-LPD that may have been caused by treatment for MDA5 antibody-positive dermatomyositis. A 53-year-old woman was diagnosed with MDA5 dermatomyositis and treated for rapidly progressive interstitial lung disease using multidrug immunosuppressive therapy with prednisolone (PSL), tacrolimus, and intravenous cyclophosphamide pulse therapy. Seven months after treatment initiation, vomiting led to the discovery of a cerebellar tumour. The cerebellar tumour was histologically Epstein-Barr virus (EBV)-encoded small RNA-positive diffuse large B-cell lymphoma, with EBV-DNA being positive in the blood. The patient was diagnosed with OIIA-LPDs due to EBV reactivation. Chemotherapy, including high-dose methotrexate (MTX) and rituximab, prevented tumour recurrence without exacerbating interstitial lung disease. This is the first reported case of CNS OIIA-LPD with multidrug immunosuppression in a patient with MDA5 dermatomyositis. Chemotherapy, including high-dose MTX and rituximab, can be used for central OIIA-LPD without aggravating settled interstitial lung disease. The activity of MDA5 dermatomyositis during OIIA-LPD treatment may be managed with low-dose PSL.