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Case Report

Atypical localization of Paget’s Disease of bone: a case series and systematic review of the literature

Franciso Bandeira, Larissa Ingrid Frazao Vasconcellos, Lilian Souza.

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INTRODUCTION: Paget's disease is characterized by altered bone remodeling and increased osteoclastic activity, with consequent changes in the structural integrity of the bone and reduction of its biomechanical efficiency, resulting in deformity, increased risk of fractures and pain. OBJECTIVES: To report four cases of Paget's disease of bone with atypical locations, as well as to carry out a systematic review of the literature in order to determined how common they are. METHODS: Case 1 – A 53 year-old male presented with bone scintigraphy showing increased MDP-Tc-99 uptake in the mandible. Case 2 – A 74 year-old male presented with pain in the left foot, plain X-ray showing sclerosis, coarse trabeculae and cortical thickening in the calcaneal bone. Case 3 – A 46 year-old female presented with pain and enlargement of the right 5th metacarpal bone which led to spontaneous fracture at the same site. MDP-Tc-99 bone scan showed increased uptake at the 5th right metacarpal, maxilla and mandible. Case 4 – A 79 year-old female presented with mandibular pain. MDP-Tc-99 bone scan showed increased uptake at maxilla, mandible and left tibia in which characteristic X-ray findings were also observed. RESULTS: Twenty- four articles were selected, totaling 30 patients with a mean age of 63.5 ± 13.6 years. The most frequently affected site was the maxilla, followed by mandible, hand bones, calcaneus and hallux. CONCLUSION: The involvement of mandible, maxilla as well as hand and foot bones are rare events in Paget's disease of bone.

Key words: Pathogenesis of periodontal disease(s), Osteoblast(s), Osteoclast(s)

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