Symptomatic hyperperfusion after superficial temporal artery-middle cerebral artery anastomosis in a child with moyamoya disease

Abstract

Object: Surgical revascularization for moyamoya disease prevents cerebral ischemic attacks by improving cerebral blood flow (CBF). It is undetermined, however, how rapid increase in CBF affects ischemic brain at acute stage, especially in children.

Case report: A 4-year-old girl with moyamoya disease underwent right superficial temporal artery-middle cerebral artery (STA-MCA) anastomosis. She suffered temporary left facial palsy 5 days after surgery. Postoperative N-isopropyl-p-[123I]iodoamphetamine single-photon emission computed tomography (123I-IMP-SPECT) revealed focal intense increase in CBF at the sites of anastomosis. Magnetic resonance imaging/angiography showed the apparently patent STA-MCA anastomosis as a thick high signal without ischemic changes. Her symptom improved 9 days after surgery, and single-photon emission computed tomography (SPECT) 2 months later showed normalization of CBF. Surgical revascularization completely relieved the transient ischemic attack on her left hand that was seen before surgery.

Conclusion: We demonstrated, for the first time, that delayed focal neurological deficit after STA-MCA anastomosis can be caused by focal hyperperfusion in childhood moyamoya disease.