Rationale:Thyroid hemangioma is benign and associated with fine-needle aspiration (FNA) biopsy or trauma in most cases. Its differential diagnosis is very difficult.Patient concerns:We presented the case of a 48-year-old man complained of slowly progressed swelling in the anterior neck for 20 years.Diagnoses:Ultrasound and CT scan revealed a hypoechogenic and heterogeneous mass measuring 4 × 3.5 cm located in the right lobe of thyroid gland. Postoperative pathological and immunohistochemical examinations of the surgical specimen revealed a primary hemangioma of the thyroid gland.Interventions:The patient received a right lobectomy of the thyroid.Outcomes:The patient had been followed up for 10 months after surgery without complications and remained asymptomatic.Lessons:Primary thyroid hemangioma should be considered when there is a well-circumscribed capsule mass on medical imaging without history of FNA or any other cervical procedures or trauma.