Kato Tomoki, Mizumoto Chisaki, Inoue Fuminori, Watanabe Takuma, Yamanaka Shigeki, Fukuhara Shizuko, Nakao Kazumasa
Department of Oral and Maxillofacial Surgery, Graduate School of Medicine, Kyoto University, Kyoto 606-8507, Japan.
Department of Hematology and Oncology, Graduate School of Medicine, Kyoto University, Kyoto 606-8507, Japan.
Exp Ther Med. 2023 Nov 28;27(1):41. doi: 10.3892/etm.2023.12329. eCollection 2024 Jan.
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.
甲氨蝶呤相关的其他医源性免疫缺陷相关淋巴增殖性疾病（MTX - OIIA - LPD）易发生结外受累，但很少累及中枢神经系统（CNS）。本研究报告了1例76岁类风湿关节炎（RA）女性患者在颌骨药物相关性骨坏死（MRONJ）治疗期间发现的CNS的MTX - OIIA - LPD病例。患者的主要诉求是右下颌磨牙处骨暴露和疼痛。该患者一直在接受用于治疗RA的甲氨蝶呤以及用于治疗骨质疏松症的阿仑膦酸钠水合物，随后使用了地诺单抗。基于MRONJ的诊断开始治疗。然而，患者随后出现头晕和飘浮感眩晕。检查发现脑部有散在的肿瘤性病变。组织病理学诊断为弥漫性大B细胞淋巴瘤。全面检查还发现肾上腺受累。由于患者正在服用甲氨蝶呤，故诊断为MTX - OIIA - LPD并停用甲氨蝶呤。由于中枢病变出现症状，开始给予化疗药物。发病8个月后，脑部和肾上腺的MTX - OIIA - LPD病变完全消退。患者身体状况改善，骨暴露区域上皮化。关于口腔颌面部区域MTX - LPD的报道很少，这可能会延迟其诊断。因此，对于正在服用甲氨蝶呤的MRONJ患者，必须对口腔病变进行活检，并与风湿病学和血液学等相关诊断科室协作，以启动口外MTX - LPD的诊断和治疗。
