Intraosseous schwannoma is extremely rare that it is not often considered among differential diagnosis for an osteolytic lesion, especially in long bones of the extremities. Amounting to less than 0.2% of all primary bone tumours and less than 200 cases reported so far, with only 3 cases involving the humerus, we hereby report the fourth case. In addition to its rarity, this was the only case of an intraosseous schwannoma involving the humerus bone which presented with a pathological fracture in a 45-year-old woman after sustaining a trivial trauma. Radiological examination revealed a geographic type of osteolytic lesion in distal shaft region of the left humerus. Only a histopathological examination helped in revealing and confirming the diagnosis of an intraosseous schwannoma. Treatment of the tumour with complete excision with bone graft reconstruction and osteosynthesis yields good results with very low risk of recurrence.
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