• Ya-Di Lu, Yen-Chen Yu, and Dun-Hao Chang.
• Department of Medical Education, Taipei Veterans General Hospital, Taipei City, Taiwan.
• Medicine (Baltimore). 2022 Oct 28; 101 (43): e30730e30730.

RationaleGranulomatous mastitis (GM) is a rare inflammatory disease and the presentation mimics infectious mastitis or breast cancer. The disease usually develops at the unilateral breast in women with breast-feeding history at their child-bearing age. Systemic steroids had been proposed as the first-line treatment, the combination of surgery was also recommended for complicated disease. However, recurrence might still happen in some rare cases. Few studies have addressed the management of such difficult situations.Patient ConcernsWe report the case of a 33-year-old androgynous and nulliparous woman who initially presented left breast erythematous swelling and was treated as infectious mastitis with debridement and antibiotics.DiagnosisAfter wider excision for pathology, the diagnosis of GM was confirmed.InterventionsSteroids combined with methotrexate were prescribed. However, the symptoms only subsided temporarily and progressed to the contralateral side within 3 months. She finally underwent double-incision mastectomy and free nipple grafting.OutcomesThe surgery was completed uneventfully, and she had a satisfactory result with no more recurrence at the 6-month follow-up.LessonThis GM case with the refractory treatment courses brought out the importance of surgical resection and was the first case report of treating GM with top surgery in the literature. Total mastectomy facilitated a highest complete remission rate of GM and may be advantageous for selected patients, especially in cases where steroids are intolerable.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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