Occipital neuralgia (ON) is commonly characterized by a neuralgiform headache accompanied by a paroxysmal burning sensation in the dermatome area of the greater, lesser, or third occipital nerve. The authors report a rare case of ON caused by an intramuscular lipoma originating from the lesser occipital nerve.A 52-year-old man presented with sharp pain in the left postauricular area with a 3 × 2-cm palpable mass. Computed tomography revealed a mass suspiciously resembling an intramuscular lipoma within splenius muscle. In the operation field, a protruding mass causing stretching of the lesser occipital nerve was found. After complete resection, the neuralgiform headache symptom had resolved and the intramuscular lipoma was confirmed through histopathology.Previous studies on the causes of ON have reported that variation in normal anatomic structures results in nerve compression. Occipital neuralgia, however, caused by intramuscular lipomas in splenius muscles have not been previously reported, and the dramatic resolution following surgery makes it an interesting case worth reporting.