Rakotoarisaona Mendrika Fifaliana, Razafimaharo Tsiory Irintsoa, Razanakoto Naina Harinjara, Sendrasoa Fandresena Arilala, Ducournau Anne, Devalland Christine, Dupond Anne-Sophie, Ranaivo Irina Mamisoa, Ramarozatovo Lala Soavina, Rapelanoro Rabenja Fahafahantsoa
Department of Dermatology, University Hospital Joseph Raseta Befelatanana, Antananarivo, Madagascar.
Department of Dermatology, Nord Franche-Comté Hospital, Trevenans, France.
Int Med Case Rep J. 2023 Mar 11;16:159-165. doi: 10.2147/IMCRJ.S403050. eCollection 2023.
Idiopathic granulomatous mastitis (IGM) is a rare chronic inflammatory disease. Neoplastic and infectious etiologies must be ruled out. IGM is a diagnostic challenge for countries with high tuberculosis endemicity like Madagascar since it may clinically and radiologically mimic breast tuberculosis. We report a case of IGM associated with erythema nodosum in a Malagasy.
A 29-year-old primiparous woman came to a dermatological consultation for typical erythema nodosum lesions that appeared one month after a breast swelling. She had no particular medical history. Examination revealed typical erythema nodosum lesions on the legs, voluminous tender mass in the right breast. Bacteriological samples and tuberculosis test were negative. Imaging showed mastitis on the right breast with no evidence of malignancy. Histology revealed a non-caseating granulomas on the lobule of the right breast. As part of an etiological work-up, COVID-19 serology was performed with a positive IgG antibody. The diagnosis of IGM associated with erythema nodosum was evocated. The evolution was favorable under systemic corticosteroid therapy.
The cause of this uncommon lesion remains obscure. The extramammary localizations such as erythema nodosum and arthralgia suggest an autoimmune origin. This pathogenesis is also reinforced by a good response to systemic immunosuppression. In our patient, the etiological assessment of the mastitis revealed a chronic infection with SARS-CoV-2. Histopathology is the gold standard for the IGM diagnosis which demonstrates a lobulocentric granulomas without caseous necrosis. Oral corticosteroid therapy is the initial choice of treatment.
Now, with several cases of concomitant IGM and EN reported, dermatologists should be aware that erythema nodosum can be one of the presenting signs of IGM, since the two conditions appear to be associated. The particularity of our case lies in the incidental discovery of SARS-CoV-2 infection. Is a chronic granulomatous disease associated with SARS-CoV-2 infection, a coincidence?
特发性肉芽肿性乳腺炎（IGM）是一种罕见的慢性炎症性疾病。必须排除肿瘤性和感染性病因。对于像马达加斯加这样结核病高流行率的国家来说，IGM是一项诊断挑战，因为它在临床和放射学上可能模仿乳腺结核。我们报告一例马达加斯加人患IGM并伴有结节性红斑的病例。
一名29岁初产妇因典型的结节性红斑皮损前来皮肤科就诊，这些皮损在乳房肿胀一个月后出现。她没有特殊病史。检查发现腿部有典型的结节性红斑皮损，右乳房有一个体积较大的压痛肿块。细菌学样本和结核菌素试验均为阴性。影像学显示右乳房有乳腺炎，无恶性肿瘤迹象。组织学检查显示右乳房小叶有非干酪样肉芽肿。作为病因学检查的一部分，进行了新冠病毒血清学检测，IgG抗体呈阳性。于是考虑诊断为IGM合并结节性红斑。在全身使用皮质类固醇治疗下病情好转。
这种罕见病变的病因仍不清楚。结节性红斑和关节痛等乳腺外定位表现提示自身免疫起源。对全身免疫抑制的良好反应也强化了这种发病机制。在我们的患者中，乳腺炎的病因评估显示为新冠病毒的慢性感染。组织病理学是IGM诊断的金标准，其显示小叶中心性肉芽肿且无干酪样坏死。口服皮质类固醇治疗是初始治疗选择。
现在，随着多例IGM和结节性红斑同时出现的病例被报道，皮肤科医生应意识到结节性红斑可能是IGM的表现体征之一，因为这两种情况似乎有关联。我们病例的特殊之处在于偶然发现了新冠病毒感染。一种与新冠病毒感染相关的慢性肉芽肿性疾病，这是巧合吗？
