Introduction: Pilonidal sinus (PNS) is any subcutaneous sinus containing hair. 97.8% of PNS are in sacrococcygeal region making it the most common site of PNS. Extrasacrococcygeal PNS accounts for approximately 2.2% of PNS. Few cases of PNS of atypical sites have been reported. The purpose of this paper is to report an unusual case of pilonidal sinus over the submandibular region. Case report: A 36 year old male patient presented with a painless swelling over left side of neck since 2 weeks which on examination had no tenderness and no local raise of temperature. Ultrasonography of neck was suggestive of abscess. Excision of the swelling was done and histopathological examination showed chronic inflammation with foreign body gaint cell reaction suggesting diagnosis of Pilonidal sinus. Conclusion: Pilonidal sinus over submandibular region is unusual and a diagnostic challenge. Total excision and primary closure was done in this case.
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