Background: Dermoid sinus disease typically arises from the congenital inclusion of ectodermal elements and can occur in various anatomical locations. Infraclavicular dermoid sinus is exceedingly rare, with very few cases documented in the literature.

Case Presentation: A 7-year-old child presented with a recurrent, purulent discharge containing hair from the right infraclavicular region for one year. Clinical examination and imaging revealed a subcutaneous sinus tract extending from 3 cm below the right clavicle down to the infraclavicular area. Surgical excision of the entire tract was performed successfully, followed by primary closure. Histopathological examination confirmed the presence of hair shafts and chronic inflammatory changes consistent with a dermoid sinus. Postoperative recovery was uneventful, with no signs of recurrence at the six-month follow-up.

Conclusion: Atypical locations of dermoid sinus can pose diagnostic dilemmas and may be mistaken for other chronic sinus or abscess formations. Complete excision of the sinus tract is necessary to prevent recurrence. This case underscores the importance of including dermoidsinus in the differential diagnosis of persistent subcutaneous sinus tracts, even in uncommon sites.

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