Introduction: Pneumosinus dilatans (PD) is a rare craniofacial malformation characterized by unusual hyperaeration of any one of the paranasal sinuses. The etiology and pathogenesis of this condition remain unclear. Here we are presenting a case study on the incidence of Pneumosinus Dilatans Frontalis (PDF) using computed tomography and in south Indian skulls. Materials and Methods: The present study was carried out in 92 dried adult human skull of unknown sex obtained from Department of Anatomy and 120 patients underwent paranasal sinus Computed tomography (CT) for various reasons at Bhasaweshwara Medical college and hospital, Chitradurga, Karnataka. The incidence of unilateral and bilateral PDF was observed and photographed. Results: Out of 90 dried skulls, the incidence of unilateral PDF was found in only one case (1.09%). In the present study, the incidence of bilateral PDF was not found in dried skulls. A total of 120 (240 slices) CT scans of the nose and paranasal sinuses were reviewed. We observed 3 cases (2.5%) of bilateral PDF among them 2 cases were seen in males and one case in female. Conclusions: PD is rare craniofacial deformity affects all paranasal sinus but most commonly affects the frontal sinus. Hence, a detailed anatomical and morphometric knowledge of the region is necessary to perform the successful surgeries and outcome in the region.