Polyostotic fibrous dysplasia (PFD) is a high-turnover bone disease that frequently entails chronic bone pain, pathological fractures and severe deformities. Recently, bisphosphonates have shown effective antiresorptive properties in the treatment of children or adults with PFD. We report on a 79-year-old female with PR), who had severe lower limb deformity and chronic bone pain in multiple sites of her extremities for more than 55 years. The patient experienced significant decrease in bone pain and bone turnover markers following long-term (8.5 years) treatment with a low-dose oral alendronate treatment (5 mg/day). To the best of our knowledge, this is the first report of a long-term follow-up of a postmenopausal elderly patient with long-standing symptomatic PFD following continuous low-dose oral alendronate therapy. This case report indicates that long-term daily administration of low-dose alendronate alone is a potential treatment option for elderly patients with PFD, particularly those with long-standing bone pain.