Paranasal sinus anatomical anomalies with unknown etiology are common. Maxillary and/or frontal sinus aplasia or hypoplasia are more common than the other types. Although there are some reports about paranasal sinus anomalies in the literature, there is no case similar to ours, with total paranasal sinus aplasia. Here, we present a unique case of total paranasal sinus aplasia with a symptom of fullness on the face.