We report here a very rare case of bilateral congenital choanal atresia untreated for the 27 years after birth. A 27-year-old Japanese man complaining of long-term nasal obstruction, snoring and daytime sleepiness visited our hospital. He exhibited the characteristic appearances of long face and bilateral hypoplastic cheeks. Examination by flexible fiberscopy and computed tomography led us to diagnose him with bilateral choanal atresia. Subsequent polysomnography revealed the number of apnoea or hypopnoea episodes per hour (apnoea-hypopnoea index, or AHI) to be over 10. On the basis of these observations, we modified the diagnosis to obstructive sleep apnoea syndrome (OSAS) secondary to congenital choanal atresia.
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