The aim of this study was to survey the association of congenitally missing anterior tooth and variations in craniofacial morphology in South Indian female patients. A total of 15 female patients with congenitally missing tooth in the maxillary anterior region were selected. 16 linear and 6 angular measurements were done on the lateral cephalograms of each subject with Legan and Burstone analysis. The cephalometric data was statistically analyzed and compared among each patient and against the cephalometric norms for the South Indian standards using Legan and Burstone analysis. The anterior hypodontia pattern showed shorter posterior cranial base length, significantly more retrognathic maxilla and prognathic mandible, flat chin, decreased upper anterior facial height, decreased mandibular angle and elongation of upper incisors. Patients with anterior hypodontia pattern have characteristic variations in craniofacial morphology. The dentofacial variations seen in persons with congenitally missing tooth is actually a functional compensation.