Noritsugu Ono, Takeshi Kusunoki, Kaori Kase, Katsuhisa Ikeda
Background: Osteomas of the nose and paranasal sinus are commonly known as benign neoplasms. Many cases with osteomas show generally asymptomatic findings.
On the other hand, some cases with manifest symptoms may require surgical treatment. The most common of these are osteomas in the paranasal sinuses, which can cause recurrent infections and headaches. It has been reported that ethmoid and frontal osteomas could extend to the orbita and cause cosmetic deformity or mucocele formation. Gardner’s syndrome is an autosomal dominant hereditary disorder characterized by clinical triad of intestinal polyposis, osteomas, and cutaneous or soft tissue tumors.
Case presentation: A 33-year-old man withan ethmoid osteoma had a past history of familial adenomatous polyposis (FAP) and a family history of FAP. The patient underwent a total colectomy due to FAP when 31 years old. His complaint was right-sided exophthalmus at the age of 33. A computed tomography demonstrated an osteoma on the roof of the anterior ethmoid region with an extension to the orbit and opacification of both maxillary and ethmoid sinuses. The osteoma could be successfully and completely removed using an endoscopic endonasal approach.
Conclusion: Suspected cases of Gardner’s syndrome in patients with paranasal sinus osteomas should have a complete workup for Gardner’s syndrome, including lower gastrointestinal tract endoscopy, barium enema imaging and genetic testing, because early recognition and diagnosis of Gardner’s syndrome will help prevent the development of colon cancer.
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