Osteonecrosis of the jaw (ONJ) is a clinical condition associated with long-term exposure to inhibitors of bone resorption, mainly bisphosphonates. Denosumab (DMab) is a human monoclonal antibody of the receptor activator of nuclear factor kappa-B ligand. It prevents osteoclast-mediated bone resorption and is widely prescribed for the management of postmenopausal osteoporosis. Whereas ONJ has already been reported in women treated with DMab, we report for the first time the development of ONJ, following tooth extraction, in a male patient treated for idiopathic osteoporosis with DMab. Due to the constant increase in DMab prescription, for the management of osteoporosis, in both genders, physicians should be made aware of this potential risk.