The authors describe a rare case of xanthogranuloma juvenile of a nose and maxillary sinus in 3 years old girl. The tumor was situated in the left nasal cavity and the left sinus. Surgical treatment was done. Histological examination disclosed xanthogranuloma juvenile. The duration of follow up was 7 months without any sight of recurrence. This report documents the first known case of maxillary sinus involvement.
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