Paranasal sinus anatomical anomalies with unknown etiology are common. Paranasal sinus-related diseases are associated with so high rate of morbidities, it becomes essential to identify the structure and pathophysiology of the paranasal sinuses. Computed tomography (CT) is a valuable tool in displaying its anatomy, any anatomic variations and diseases. As paranasal sinus development is a complex and long-lasting process, there are great structural variations between individuals. Maxillary and/or frontal sinus aplasia or hypoplasia are more common than the other types. Several degrees and combinations of aplasias and hypoplasias have been reported; however, there is only two case of total paranasal sinus aplasia in the literature. Here, we present the third case of total paranasal sinus aplasia, and the first ever case reported from India.