Department of Rheumatology and Immunology, Changde Hospital Affiliated to Xiangya School of Medicine, Central South University & First People's Hospital of Changde City, Changde Hunan 415003, China.
Zhong Nan Da Xue Xue Bao Yi Xue Ban. 2024 May 28;49(5):818-824. doi: 10.11817/j.issn.1672-7347.2024.230501.
Actinomycosis is a rare chronic granulomatous disease characterized by granuloma formation and tissue fibrosis with sinus tracts, often misdiagnosed due to its similarity to many infectious and non-infectious diseases. This report presents a case of a 60-year-old female with more than 10 years history of rheumatoid arthritis who developed actinomycosis infection after long-term treatment with immunosuppressants and biologics, including methotrexate, leflunomide, and infliximab, leading to recurrent joint pain, poorly controlled rheumatoid arthritis activity, and persistent elevation of white blood cell counts. Abdominal CT revealed a pelvic mass and right ureteral dilation. Pathological examination of cervical tissue showed significant neutrophil infiltration and sulfur granules, indicating actinomycosis. The patient received 18 months of doxycycline treatment for the infection and continued rheumatoid arthritis therapy with leflunomide, hydroxychloroquine sulfate, and tofacitinib, resulting in improved joint symptoms and normalized white blood cell counts. After 2 years of follow-up, the patient remained stable with no recurrence. This case highlights the importance of clinicians being vigilant for infections, particularly chronic, occult infections from rare pathogens, in rheumatoid arthritis patients on potent immunosuppressants and biologics, advocating for early screening and diagnosis.
放线菌病是一种罕见的慢性肉芽肿性疾病,其特征为肉芽肿形成和组织纤维化伴窦道形成,由于其与许多感染性和非感染性疾病相似,常被误诊。本报告介绍了一例 60 岁女性患者,患有类风湿关节炎 10 余年,长期接受免疫抑制剂和生物制剂(包括甲氨蝶呤、来氟米特和英夫利昔单抗)治疗后发生放线菌感染,导致反复关节疼痛、类风湿关节炎活动度控制不佳和白细胞计数持续升高。腹部 CT 显示骨盆肿块和右侧输尿管扩张。宫颈组织的病理检查显示明显的中性粒细胞浸润和硫磺颗粒,提示放线菌病。患者接受了 18 个月的多西环素治疗感染,并继续接受来氟米特、硫酸羟氯喹和托法替尼治疗类风湿关节炎,关节症状得到改善,白细胞计数恢复正常。随访 2 年后,患者病情稳定,无复发。本病例强调了临床医生在使用强效免疫抑制剂和生物制剂治疗类风湿关节炎患者时对感染,特别是来自罕见病原体的慢性隐匿性感染保持警惕的重要性,倡导早期筛查和诊断。