Division of Arthritis and Rheumatic Diseases, Department of Medicine, Oregon Health & Science University, Portland, OR, USA.
Int J Rheum Dis. 2021 Apr;24(4):526-532. doi: 10.1111/1756-185X.14065. Epub 2021 Feb 1.
Idiopathic granulomatous mastitis (IGM) is an enigmatic inflammatory breast disorder. IGM responds to immunomodulatory treatment and may be associated with systemic manifestations such as arthritis and erythema nodosum. These patients are increasingly referred to rheumatologists for management, but IGM is rarely discussed in the rheumatology literature. The objective of this report is to familiarize rheumatologists with the treatment and systemic manifestations of IGM. We report here a case series of IGM at our institution, and a literature review of IGM treated with methotrexate (MTX).
Patients with IGM at our institution were identified and described using a retrospective chart review. A literature review of PubMed and Google Scholar identified studies of IGM patients treated with MTX.
We identified 28 IGM patients at our institution. Inflammatory arthritis/arthralgia were present in four patients (14%), and five patients (18%) had erythema nodosum. Patients treated with MTX had the highest rates of relapse-free remission; relapse-free remission occurred in four of the five (80%) MTX-treated patients, compared with 5 of 12 (42%) patients treated with steroids alone, and two or three (66%) patients treated with steroids and surgery. In the literature review, 116 patients treated with MTX were identified, and the rate of relapse-free remission ranged from 58% to 100%. Arthritis/arthralgia and erythema nodosum were more common at our institution than reported in the literature.
Methotrexate is a promising treatment for IGM. Arthritis/arthralgias and erythema nodosum may be under-recognized when IGM patients are managed outside rheumatology. Prospective studies are needed to characterize clinical features and optimum treatment of IGM.
特发性肉芽肿性乳腺炎（IGM）是一种神秘的炎症性乳腺疾病。IGM 对免疫调节治疗有反应，并且可能与关节炎和结节性红斑等全身表现相关。这些患者越来越多地被转介给风湿病医生进行管理，但 IGM 在风湿病文献中很少被讨论。本报告的目的是使风湿病医生熟悉 IGM 的治疗和全身表现。我们在此报告我们机构的 IGM 病例系列，并对接受甲氨蝶呤（MTX）治疗的 IGM 进行文献复习。
通过回顾性病历回顾，确定并描述我们机构的 IGM 患者。通过 PubMed 和 Google Scholar 对 IGM 患者接受 MTX 治疗的研究进行文献复习。
我们在我们的机构中确定了 28 例 IGM 患者。四名患者（14%）存在炎性关节炎/关节痛，五名患者（18%）存在结节性红斑。接受 MTX 治疗的患者无复发缓解率最高；五名接受 MTX 治疗的患者中有四名（80%）无复发缓解，而仅接受类固醇治疗的 12 名患者中有 5 名（42%），接受类固醇和手术治疗的患者中有两或三名（66%）。在文献复习中，确定了 116 例接受 MTX 治疗的患者，无复发缓解率范围为 58%至 100%。在我们的机构中，关节炎/关节痛和结节性红斑比文献报道更常见。
甲氨蝶呤是 IGM 的一种有前途的治疗方法。当 IGM 患者在风湿病学之外进行管理时，关节炎/关节痛和结节性红斑可能被低估。需要前瞻性研究来描述 IGM 的临床特征和最佳治疗方法。
