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Klipple-Trenaunay syndrome (KTS) is characterized by three symptoms including hemangioma, varicose veins and hypertrophy of bony and soft tissues. Hypertrophy usually appears unilaterally, and mostly involves lower limb, however, trunk, upper limb, and face may also be affected. The occurrence of orofacial manifestations in KTS is rare. In this article a case with hemifacial hypertrophy as a rare manifestation of KTS who underwent surgery based on patient demand of aesthetic has been presented. Further, presentations of KTS in head and neck region and considerations for corrective surgery of these patients have been discussed.