The extremely rare localization of an intramuscular hemangioma (IMH) into the anterior scalene muscle was the motive for the present case report, aiming to highlight major, atypical characteristics. An 11-month-old boy with free medical history presented with a painless and progressively growing lesion 4.5 × 4 cm in diameter, located in the left supraclavicular region over the last 4 months. During physical examination, the presence of a painless, non-pulsating, non-adhesive to the overlying skin lesion was documented. Color Doppler flow ultrasonographic examination demonstrated the increased blood supply to the aforementioned lesion. Thus, we planned an elective surgical excision of the lesion in healthy limits. The postoperative course was uneventful, and the patient was discharged on the second postoperative day in good general condition. Histopathologic examination revealed the presence of hemangioma surrounded by connective tissue bundles and striated muscle fibers. IMHs do not follow the general rule of regression, beyond the age of 6–12 months, with no trend to increase over time. Accurate preoperative diagnosis is challenging. Color Doppler flow ultrasonographic examination is the imaging modality of choice during the preoperative assessment. Surgical excision of the IMH in healthy limits is the most appropriate treatment option.