Objective
Granulomatous 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.
Case presentation
Herein, we describe the case of a 33-year-old androgynous and nulliparous woman with bilateral refractory GM. She initially presented left breast erythematous swelling and was treated as infectious mastitis with debridement and antibiotics. After wider excision for pathology, the diagnosis of GM was confirmed. Steroids combined with methotrexate were prescribed. However, the symptoms only subsided temporarily and progressed to the contralateral side within 3 months. Finally, she underwent double-incision mastectomy and free nipple grafting and had a satisfactory result with no more recurrence at the 6-month follow-up.
Conclusion
This 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.

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