Case Presentation: A 16-year-old female presented with complaints of serous discharge mixed with pus intermittently from the intermammary region for 5 months. The patient was treated successfully with excision and primary closure.

Discussion: Although a pilonidal sinus is most commonly seen at the sacro-coccygeal region, it can occur occasionally at various other locations in the body. The presence of one such entity in the intermammary region of a female represents a rare occurrence with a very few cases reported worldwide. A proper surgical repair in terms of its excision and closure remains the mainstay in management.

Conclusion: An intermammary pilonidal sinus is an extremely rare condition seen in young, obese females with large, pendulous and heavy breasts.