{"title":"与甲氨蝶呤相关的中枢神经系统其他先天性免疫缺陷相关淋巴组织增生性疾病和与药物相关的颌骨坏死同时发生:病例报告。","authors":"Tomoki Kato, Chisaki Mizumoto, Fuminori Inoue, Takuma Watanabe, Shigeki Yamanaka, Shizuko Fukuhara, Kazumasa Nakao","doi":"10.3892/etm.2023.12329","DOIUrl":null,"url":null,"abstract":"Methotrexate-related other iatrogenic immunodeficiency-associated lymphoproliferative disorder (MTX-OIIA-LPD) is prone to extranodal involvement but rarely involves the central nervous system (CNS). The present study reports a case of MTX-OIIA-LPD of the CNS discovered during medication-related osteonecrosis of the jaw (MRONJ) treatment in a 76-year-old woman with rheumatoid arthritis (RA). The chief complaint of the patient was bone exposure and pain in the right mandibular molar. The patient had been receiving MTX for RA and alendronate sodium hydrate for osteoporosis, followed by denosumab. Treatment was initiated based on a diagnosis of MRONJ. However, the patient experienced lightheadedness and floating dizziness afterwards. Examinations revealed scattered neoplastic lesions in the brain. The histopathological diagnosis was diffuse large B-cell lymphoma. A systemic search also revealed adrenal involvement. Since the patient was taking MTX, a diagnosis of MTX-OIIA-LPD was made and MTX was discontinued. Chemotherapeutic agents were administered since the central lesions became symptomatic. The MTX-OIIA-LPD lesions in the brain and adrenal glands completely resolved 8 months after onset. The physical condition of the patient improved, and the bone-exposed areas became epithelialized. Reports on MTX-LPD in the oral and maxillofacial region are few, which may delay its diagnosis. Therefore, biopsy of oral lesions in patients with MRONJ who are taking MTX and collaboration with related diagnostic departments, such as rheumatology and hematology, must be done to initiate the diagnosis and treatment of extraoral MTX-LPD.","PeriodicalId":12285,"journal":{"name":"Experimental and therapeutic medicine","volume":"14 1","pages":""},"PeriodicalIF":2.4000,"publicationDate":"2023-11-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Methotrexate‑related other iatrogenic immunodeficiency‑associated lymphoproliferative disorder in the CNS and medication‑related osteonecrosis of the jaw occurring simultaneously: A case report.\",\"authors\":\"Tomoki Kato, Chisaki Mizumoto, Fuminori Inoue, Takuma Watanabe, Shigeki Yamanaka, Shizuko Fukuhara, Kazumasa Nakao\",\"doi\":\"10.3892/etm.2023.12329\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"Methotrexate-related other iatrogenic immunodeficiency-associated lymphoproliferative disorder (MTX-OIIA-LPD) is prone to extranodal involvement but rarely involves the central nervous system (CNS). The present study reports a case of MTX-OIIA-LPD of the CNS discovered during medication-related osteonecrosis of the jaw (MRONJ) treatment in a 76-year-old woman with rheumatoid arthritis (RA). The chief complaint of the patient was bone exposure and pain in the right mandibular molar. The patient had been receiving MTX for RA and alendronate sodium hydrate for osteoporosis, followed by denosumab. Treatment was initiated based on a diagnosis of MRONJ. However, the patient experienced lightheadedness and floating dizziness afterwards. Examinations revealed scattered neoplastic lesions in the brain. The histopathological diagnosis was diffuse large B-cell lymphoma. A systemic search also revealed adrenal involvement. Since the patient was taking MTX, a diagnosis of MTX-OIIA-LPD was made and MTX was discontinued. Chemotherapeutic agents were administered since the central lesions became symptomatic. The MTX-OIIA-LPD lesions in the brain and adrenal glands completely resolved 8 months after onset. The physical condition of the patient improved, and the bone-exposed areas became epithelialized. Reports on MTX-LPD in the oral and maxillofacial region are few, which may delay its diagnosis. Therefore, biopsy of oral lesions in patients with MRONJ who are taking MTX and collaboration with related diagnostic departments, such as rheumatology and hematology, must be done to initiate the diagnosis and treatment of extraoral MTX-LPD.\",\"PeriodicalId\":12285,\"journal\":{\"name\":\"Experimental and therapeutic medicine\",\"volume\":\"14 1\",\"pages\":\"\"},\"PeriodicalIF\":2.4000,\"publicationDate\":\"2023-11-28\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Experimental and therapeutic medicine\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.3892/etm.2023.12329\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"MEDICINE, RESEARCH & EXPERIMENTAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Experimental and therapeutic medicine","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.3892/etm.2023.12329","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"MEDICINE, RESEARCH & EXPERIMENTAL","Score":null,"Total":0}
引用次数: 0
摘要
甲氨蝶呤相关的其他先天性免疫缺陷相关淋巴组织增生性疾病(MTX-OIIA-LPD)容易累及结节外,但很少累及中枢神经系统(CNS)。本研究报告了一例中枢神经系统MTX-OIIA-LPD病例,患者是一名76岁的类风湿性关节炎(RA)女性患者,在接受药物相关性颌骨坏死(MRONJ)治疗期间被发现。患者的主诉是右下颌臼齿骨质暴露和疼痛。患者一直在接受 MTX 治疗 RA 和阿仑膦酸钠水合物治疗骨质疏松症,之后又接受了地诺单抗治疗。治疗是根据 MRONJ 诊断开始的。然而,患者随后出现了头晕和漂浮感。检查发现脑部有散在的肿瘤病变。组织病理学诊断为弥漫大 B 细胞淋巴瘤。系统检查还发现肾上腺受累。由于患者正在服用MTX,因此诊断为MTX-OIIA-LPD,并停用了MTX。由于中心病变已出现症状,因此使用了化疗药物。脑部和肾上腺的 MTX-OIIA-LPD 病变在发病 8 个月后完全消退。患者的身体状况有所改善,骨头暴露部位也上了一层皮。有关口腔和颌面部 MTX-LPD 的报道很少,这可能会延误诊断。因此,必须对正在服用MTX的MRONJ患者的口腔病变进行活检,并与风湿病学和血液学等相关诊断部门合作,以启动口腔外MTX-LPD的诊断和治疗。
Methotrexate‑related other iatrogenic immunodeficiency‑associated lymphoproliferative disorder in the CNS and medication‑related osteonecrosis of the jaw occurring simultaneously: A case report.
Methotrexate-related other iatrogenic immunodeficiency-associated lymphoproliferative disorder (MTX-OIIA-LPD) is prone to extranodal involvement but rarely involves the central nervous system (CNS). The present study reports a case of MTX-OIIA-LPD of the CNS discovered during medication-related osteonecrosis of the jaw (MRONJ) treatment in a 76-year-old woman with rheumatoid arthritis (RA). The chief complaint of the patient was bone exposure and pain in the right mandibular molar. The patient had been receiving MTX for RA and alendronate sodium hydrate for osteoporosis, followed by denosumab. Treatment was initiated based on a diagnosis of MRONJ. However, the patient experienced lightheadedness and floating dizziness afterwards. Examinations revealed scattered neoplastic lesions in the brain. The histopathological diagnosis was diffuse large B-cell lymphoma. A systemic search also revealed adrenal involvement. Since the patient was taking MTX, a diagnosis of MTX-OIIA-LPD was made and MTX was discontinued. Chemotherapeutic agents were administered since the central lesions became symptomatic. The MTX-OIIA-LPD lesions in the brain and adrenal glands completely resolved 8 months after onset. The physical condition of the patient improved, and the bone-exposed areas became epithelialized. Reports on MTX-LPD in the oral and maxillofacial region are few, which may delay its diagnosis. Therefore, biopsy of oral lesions in patients with MRONJ who are taking MTX and collaboration with related diagnostic departments, such as rheumatology and hematology, must be done to initiate the diagnosis and treatment of extraoral MTX-LPD.