• Rong Shi, Peng Liu, and Xuan-Fen Zhang.
• Department of Mammary Glands, Gansu Provincial Hospital.
• Medicine (Baltimore). 2021 Mar 26; 100 (12): e25166e25166.

RationalePilonidal sinus disease (PSD) involving the breast is extremely rare and has not been described in man.Patient ConcernsThis current case report presents a case of a pilonidal cyst in a 46-year-old man which was surgically treated. He had intermittent pain in his left breast for 2 months and came for local rupture and discharge for 1 week.DiagnosisThe initial diagnosis is male mastitis, on the basis of the histological features of H&E-stained specimens and immunohistochemistry of the resected lump, this case was diagnosed as PSD.InterventionsThe patient underwent "enlarged resection of the left breast lesion" under local anesthesia.OutcomesThe patient's surgical area healed well, without any signs of recurrence.ConclusionPSD involving the breast is extremely rare in man, with no typically clinical manifestations, and could be easily ignored. This disease requires great attentions from clinicians.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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