Osteoid osteoma is a common benign primary bone tumor, but it is very uncommon in the proximal humerus. This case report describes the clinical course and treatment of a patient with shoulder pain and osteoid osteoma of the proximal humerus and provides a review of the literature. A 22-year-old healthy male patient presented to our clinic with a 2-year history of constant throbbing right shoulder pain. The patient was referred for orthopedic consultation. A series of plain radiographs, bone scintigraphy, and a magnetic resonance imaging were done and revealed an osseous lesion at the medial aspect of the proximal meta diaphyseal region of the right proximal humerus, with a diagnosis of osteoid osteoma. The patient underwent radiofrequency ablation of the tumor nidus, which was successful and resulted in resolution of symptoms with minimal pain at follow up. This case demonstrates that osteoid osteoma can present with clinical features that mimic various causes for shoulder pain.