A case of autosomal dominant osteosclerosis in a 44-year-old woman is presented. Hyperostosis of the maxillo-mandibular alveolus is featured in clinical symptom through the dent-maxillofacial examination. Routine radiografic examination revealed the findings of osteosclerosis and hyperostosis of the jaw, endosteal sclerosis of the neurocranium with loss of the diploe, and also endosteal sclerosis of the diaphyses of tubular bones. Laboratory data shows only the serum 1, 25 (OH) 2 vitamin D3 level is in higher abnormal limit. Bone biopsy taken from the iliac crest proved the histopathological findings of the narrowing medullary cavity as well as the widened, increased amounts of travecular bone, and significantly decreased osteoblast and osteoclast.
