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Infantile osteopetrosis associated with osteomyelitis. Osteopetrosis is a rare inherited disorder characterised by the inability to reabsorb and remodel bone due to an osteoclast dysfunction. An increase in bone mass and density results in severe skeletal malformation and bone marrow failure, which may be fatal. The aim of this study was to report a case of infantile osteopetrosis in a 6-year-old boy, who primarily complained of dental problems. The patient developed an osteomyelitis after a primary tooth extraction. We also reviewed the literature about this genetic bone disease, since the management of dental problems of these patients can be a challenge to the dentist. The diagnosis, clinical and radiographic features and treatment of the case were discussed in this work.