In this study, an unusual case of osteoma is presented, whereby a bone marrow osteoma was identified in the tibia. No previous cases of bone marrow osteoma have been reported. In this case, an eight-year-old male presented with discontinuous discomfort in the right distal calf for six months. Radiological examination and computed tomography revealed a radiopaque lesion within the affected bone. A technetium-99m bone scan revealed focally increased uptake in the same region. Together, these observations prior to surgery indicated that the patient may suffer from bone disease. Subsequently, a surgical excision was performed and the biopsy specimen was identified as bone marrow osteoma. Following surgery, the symptoms were eradicated and the prognosis was positive during the 24-month follow-up period. Bone marrow osteoma should be considered when a patient suffers from discontinuous and unexplained limb discomfort.