The authors report a case of mycotic osteitis of the upper maxillary bone due to Candida Albicans occurring in a 56 year old female patient under treatment for chronic myeloid leukemia. The etiologically difficult diagnosis could only be confirmed after deep surgical biopsy with mycological study of a fragment. A review of the literature confirmed the rarity of upper maxillary involvement by Candida Albicans. Particularly when there is isolated involvement and no evidence of a distant primary focus. The differential diagnosis essentially includes centro-facial malignant granuloma.