Abstract: Bone islands are completely benign bone lesions, most of the bone islands are small. In clinical, bone imaging shows that bone island with increased metabolism is rare. The author reported a case of bone island with ⁹⁹Tc^m-methylenediphosphonate(MDP) SPECT/CT showing increased metabolism. The characteristics of the disease were analyzed including the clinical symptoms, imaging manifestations, pathological diagnosis and histopathology, and were deepened the understanding through literature review. The aim of this study is to improve the awareness of the disease and to avoid misdiagnosis for imaging physicians.