Case of bone Paget's disease with cheek pain, hearing loss and tremor whose histological findings showed peculiar calcified structures

An 85-year-old man visited our department of oral surgery and dentistry with a chief complaint of right cheek pain. He had suffered from hearing loss, head enlargement, involuntary facial movement, and kinetic tremor that had progressed over 20 years. Laboratory examination revealed an extremely high level of serum alkaline phosphatase (5581 IU/L) and bone turnover markers. Skull X-rays showed “cotton-wool” appearance and multiple cysts were also recognized with computed tomography. The histological findings of the bone biopsy specimen supported the diagnosis of bone Paget's disease but peculiar spheroid calcified structures were observed. Oral administration of alendronate (5 mg/day) was tolerated and the bone turnover was successfully controlled.