Superficial temporal artery-middle cerebral artery (STA-MCA) anastomosis is a standard surgical therapeutic option in patients with moyamoya disease. Most patients experience improvement in their clinical symptoms immediately after surgery. The authors report on the case of a 39-year-old man with moyamoya disease who suffered from temporary and frequent neurological deterioration after undergoing a left STA-MCA anastomosis. Hemodilution and hypervolemia therapies did not improve his course. Technetium-99m hexamethylpropyleneamine oxime single-photon emission tomography scans demonstrated focal intense accumulation of the tracer in the frontal operculum on the side of the surgery. Although diffusion-weighted magnetic resonance (MR) imaging demonstrated no abnormalities except for the postoperative change, perfusion-weighted MR images and MR digital subtraction angiography revealed focal hyperperfusion in the left frontal operculum where the cerebral perfusion reserve was severely disturbed preoperatively. This evidence strongly supports the notion that focal hyperperfusion observed after STA-MCA anastomosis could occur in the poor perfusion reserve area preoperatively and could cause temporary neurological deterioration.