Intriduction: A cholesteatoma is a cystic keratin-filled mass lined with stratified squamous epithelium. Paranasal sinus cholesteatoma is a rare clinical entity with only a few cases reported in the literature. Case Report: We describe a rare case of cholesteatoma in the maxillary and ethmoid sinus presenting with blood-stained nasal discharge and nasal obstruction refractory to medical treatment. The patient was treated by functional endoscopic sinus surgery to remove the cholesteatoma completely. Final Consideration: Through the results obtained in this study, corroborates that it is necessaryto create a large antrostomy to ensure completely removing the cholesteatoma and facilitate to easier follow-up.