Cleft lip and palate is the most commonly reported congenital anomaly requiring early surgery, occurring at a rate of 79.1 per 100,000 live births. Hypertrophic scar formation is a postoperative main complication of cleft lip and palate repair and may often surgical revision. The pathogenesis of hypertrophic scars is not clear and much of understanding is from anecdotal, clinical experience. Hypertrophic scars formation can result in esthetically deforming scars that may lead to secondary cleft lip revision. Aberrant scar formation following cleft lip repair is associated with lip asymmetry and nasal stenosis. This case study reported unusual severe nasal stenosis associated with aberrant hypertrophic scar in an incomplete unilateral cleft lip patient and reviewed relevant articles