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A case of extensive ossification around the left hip joint involving lesser trochanter of the femur leading to ankylosis of left hip joint in a 60 years male is being reported. The diagnosis of moyamoya disease was made, which is a rare form of occlusive cerebrovascular disorder. Occlusion of an artery may present with Transient Ischemic Attacks, headaches, stroke and seizures. Surgical management is the only option in these cases. This 60 years male had an acute onset hemiplegia 4½ years back and at present came with complaints of pain and swelling over the left hip. Surgical excision was done and histopathological examination revealed extensive ossification of skeletal muscle. A diagnosis of Moyamoya disease complicated with heterotopic ossification was made. There was no recurrence after 8 months of follow up.