The paranasal sinuses are air filled spaces. The process of development of paranasal sinuses begins prenatally. The agenesis of paranasal sinuses in an unusual clinical condition and that is mainly confined to the frontal sinus unilaterally. Combined aplasia of multiple sinuses is extremely rare along with hypoplasia of other sinuses. Agenesis of sphenoid sinus is also rare in incidence. We report a case of a 54 years old female, who presented with complains of persistent headache with nasal blockage and found to have combined aplasia of bilateral frontal and sphenoid sinus with hypoplasia of bilateral maxillary and ethmoid sinus with unusual age of presentation. The findings were confirmed on non-contrast computed tomography scan of paranasal sinuses.
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