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Highlights • Sacrococcygeal PNS is the commonest variant of the condition. • However, it can occur in other areas like umbilicus, hand, scalp intermammary, suprapubic, nose, etc. • Infra mammary PNS is an extremely rare condition, with no previous report. • This study aims to present and discuss a case of PNS occurring in both infra mammary regions., Introduction Pilonidal sinus (PNS) is a chronic inflammatory perianal disorder that rarely occurs outside sacrococcygeal region. The aim of this study is to report an extremely rare case of bilateral inframammary PNS with brief literature review. Case report A 25-year-old female presented with a discharging sinuses in both inframammary region for two years. Examination showed multiple discharging sinuses with several centimeters of induration and tenderness. Under general anesthesia, complete excision of the sinuses with primary closure done. Histopathological examinations showed chronic foreign body granuloma surrounding hair shaft pictures consistent with PNS. Discussion Inframammary PNS has never been reported in the literature. As with intermammary PNS, in this case also it is associated with obesity and large breasts with tight brassieres. Diagnosis is usually clinical. In contrast to sacrococcygeal PNS, operation under general anesthesia is main treatment modality. Conclusion Pilonidal sinus of inframammary region is an extremely rare condition. High index of suspicion is required for diagnosis. Excision with primary closure is the definitive therapy.