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Spinal angiolipoma is a benign lesion and presents with compressive myelopathy. Typically, it is located in epidural compartment. However, intramedullary angiolipoma is extremely uncommon, and till date only eight cases are reported in the literature and all reported cases had isolated solitary lesion. Authors report an interesting case of intramedullary spinal angiolipoma (ISAL) in a - 48- years male, presented with compressive myelopathy, magnetic resonance imaging study revealed presence of two separate angiolipomas, which were located at D8-D9 and D10-D12 vertebral levels respectively, underwent successful near total surgical resection with good neurological outcome. Current case represents first of its kind in the western literature. Management of such rare pathology along with pertinent literature is briefly discussed.