Bilateral congenital choanal atresia and osteoma of ethmoid sinus with supernumerary nostril: a case report and review of the literature

Xue-Zhong Li,Xiao-Lan Cai,Xiao Wei,Lei Zhang,Xue-Feng Han

doi:10.1186/1752-1947-5-583

Xue-Zhong Li, Xiao-Lan Cai + Show 3 more

Open Access

https://doi.org/10.1186/1752-1947-5-583

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Abstract

IntroductionCongenital choanal atresia is a relatively rare deformity, especially bilateral congenital choanal atresia. We report a case of bilateral congenital choanal atresia in a 22-year-old Chinese man, who was also diagnosed with congenital right accessory nasal deformity, osteoma of his left ethmoid sinus and congenital keratoleukoma of his right eye.Case presentationA 22-year-old Chinese man presented with mouth breathing, sleep snoring and difficult feeding after birth, with no olfactory sensation. Three-dimensional computed tomography revealed bilateral choanal atresia and a high density bony shadow in his left ethmoid sinus that extended to his left frontal sinus.ConclusionsChoanal atresia is often accompanied by other congenital abnormalities. To the best of our knowledge, this is the first report of choanal atresia accompanied by congenital accessory nasal deformity and congenital keratoleukoma.

Highlights

Congenital choanal atresia is a relatively rare deformity, especially bilateral congenital choanal atresia
Choanal atresia is often accompanied by other congenital abnormalities
To the best of our knowledge, this is the first report of choanal atresia accompanied by congenital accessory nasal deformity and congenital keratoleukoma

Summary

Background

Choanal atresia was first reported by Johann Roderer in 1755 [1]. Its incidence is about 8.2 per 100, 000 [2] and the primary symptoms, such as bilateral imperforation, dyspnea and inability to suck after birth, are attributed to postnatal nasal obstruction. We present a rare case of bilateral congenital choanal atresia, which was accompanied by congenital right accessory nasal deformity, osteoma of his ethmoid sinus, and congenital keratoleukoma of his right eye. Our primary diagnosis was congenital right accessory nasal deformity (Figure 1), congenital bilateral atresia of the posterior nares (Figures 2 and 5), osteoma of the left ethmoid sinus (Figure 3) and congenital keratoleukoma of his right eye (Figure 4). He was operated on under general anesthesia in March 2007.

Discussion

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Conclusion