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Lipoblastoma is a rare, benign, and encapsulated tumor arising from embryonic white fat. Most of the cases occur in the extremities and the trunk; only a few cases in the head and the neck are reported. Thus, we present a case of lipoblastoma of the neck with a review of the literature.A 1-year-old male infant presented to our hospital, with a history of painless swelling in the left side of the neck for 3 months that was rapidly enlarged. His birth history and medical history were unremarkable. A physical examination demonstrated a soft and compressible mass in the left side of the neck. The mass was nontender to palpation and mobile without cellulitic changes in the overlying skin. A computed tomographic scan showed that the mass is heterogenous, has low attenuation in nature, and is 3.8 × 2.8 × 9 cm in size.Under general anesthesia, transverse cervical incision was made through the neck wrinkle, and there was no invasion of any of the neck structures. Complete surgical excision demonstrated yellowish-white, irregular lobules of immature fat cells separated by a loose and myxoid connective tissue. Grossly, the mass was a homogeneous tan-pink gelatinous mass. A microscopic examination demonstrated a small number of capillaries and mature fat cells, and differentiating immature lipoblastoma cells were detected in the myxoid stroma. A pathologic finding confirmed the diagnosis of lipoblastoma. The postoperative course was uneventful. The patient underwent follow-up for 1 year after the operation, and there was no evidence of recurrence.The most common presentation of lipoblastoma is a painless, rapidly enlarging neck mass. Published reports showed that most of them occur before the age of 3 years. Complete surgical excision is the treatment of choice. Although lipoblastoma is an extremely rare benign tumor, it should be considered in the diagnosis of neck mass in children younger than 3 years.