<p>Branchial anomalies are composed of a heterogeneous group of congenital malformations that arise from incomplete obliteration of pharyngeal clefts and pouches during embryogenesis. Although present at birth, many abnormalities do not become evident until later in infancy or childhood. A 6-year-old male patient has rare site of opening of first branchial sinus opening on face. CT sinogram with contrast showed the tract and dissection was carried out under general anaesthesia following which patient had uneventful recovery. This case report shows such rare presentation of the first branchial cleft sinus opening on face. Due to its rare location on face and limited literature, it posed diagnostic challenges and inadequate treatment.</p>