Osteoid osteoma, a frequent lesions of bone, is usually intraosseous but occasionally subperiosteal. We describe the case of a 19-year-old male with knee pain caused by subperiosteal osteoid osteoma. Radiologic evaluation was performed with radiographic, computed tomography (CT), ultrasonographic (US) and magnetic resonance imaging (MRI). But the preoperative diagnosis of osteoid osteoma was delayed because of unusual imaging findings and atypical symptom. After excisional biopsy, histological examination confirmed the diagnosis of osteoid osteoma. The lesion was treated successfully with CT-guided radiofrequency ablation.