Rationale and objectives: Although computed tomography (CT) guided radiofrequency ablation (RFA) is accepted as the gold standard treatment for osteoid osteoma (OO) in extremities, most researchers have omitted OO in critical sites. We aimed to evaluate the efficacy and safety of CT-guided RFA in the management of OO in atypical sites. Materials and methods: A prospective interventional study conducted on 34 patients (12 women and 22 men; mean age, 22.3 ± 3.5 years; range, 12-48 years) with OO in atypical sites. All patients were treated with CT-guided RFA. All procedures were performed under general anesthesia using a single straight, rigid RF electrode for 6 minutes at 90°C. All patients were followed up; technical and clinical successes of treatment were evaluated. The severity of pain was assessed on a visual analogue scale (VAS). Results: CT-guided RFA was performed in all patients with 100% technical success and 100% primary clinical success. All patients had complete pain relief and returned to their quality of life without further analgesics within 1 month after the procedure. All procedures were successfully completed without any major complications. The mean preprocedure VAS score dropped to zero by the end of the first week with a highly statistically significant difference (p < 0.001). Conclusion: CT-guided RFA is a safe and effective procedure in the management of OO in atypical sites with 100% primary success rate, 0% long-term recurrence rate, and very low complication rate.