A case of angiomyolipoma found in the subgaleal lesion is reported. Lipoma appearing between the galea and periosteum of the frontal bone is called subgaleal or subfascial lipoma and usually consists histopathologically of mature fat cells. We experienced a case of so-called subgaleal lipoma; however, the distinctive histologic features of our case were the proliferation of non-striated muscle fibers and small vessels scattered throughout the mature fat cells, which were compatible with angiomyolipoma. Extrarenal angiomyolipoma, especially cutaneous angiomyolipoma, are very rarely mentioned in the literature. Our case is the second reported patient with angiomyolipoma in the subgaleal location.