Intramuscular hemangioma (IH) is relatively uncommon, accounting for less than 1% of all hemangiomas, usually located within the skeletal muscles of the trunk or limbs, particularly in the pelvic region. Approximately 10% of IH may occur in the head and neck region, most commonly affecting the masseter, sternocleidomastoid, and trapezius muscles. A 57-year-old man was referred for evaluation of a 4-cm asymptomatic, bluish, and slow-growing swelling of the lower lip. The patient's history included lower lip trauma and bleeding 5 years earlier. The clinical differential diagnosis included mucocele, angioleiomyoma, and salivary gland tumor. The patient was submitted to excisional biopsy under general anesthesia. Microscopically, proliferating vascular channels lined by endothelium and smooth muscle were observed infiltrating diffusely skeletal muscles, adipose tissue, and connective tissue. The final diagnosis was IH. The patient is under regular follow-up with no recurrence. IH rarely affects the lips. Intramuscular hemangioma (IH) is relatively uncommon, accounting for less than 1% of all hemangiomas, usually located within the skeletal muscles of the trunk or limbs, particularly in the pelvic region. Approximately 10% of IH may occur in the head and neck region, most commonly affecting the masseter, sternocleidomastoid, and trapezius muscles. A 57-year-old man was referred for evaluation of a 4-cm asymptomatic, bluish, and slow-growing swelling of the lower lip. The patient's history included lower lip trauma and bleeding 5 years earlier. The clinical differential diagnosis included mucocele, angioleiomyoma, and salivary gland tumor. The patient was submitted to excisional biopsy under general anesthesia. Microscopically, proliferating vascular channels lined by endothelium and smooth muscle were observed infiltrating diffusely skeletal muscles, adipose tissue, and connective tissue. The final diagnosis was IH. The patient is under regular follow-up with no recurrence. IH rarely affects the lips.