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We performed positron emission tomographic imaging with Pittsburgh Compound B on 42\u2010year\u2010old man with early manifestations of Iowa\u2010type hereditary cerebral amyloid angiopathy, a form of the disorder with little or no plaque deposits of fibrillar \u03b2\u2010amyloid. The results demonstrated increased Pittsburgh Compound B retention selectively in occipital cortex, sparing regions typically labeled in Alzheimer's disease. These results offer compelling evidence that Pittsburgh Compound B positron emission tomography can noninvasively detect isolated cerebral amyloid angiopathy before overt signs of tissue damage such as hemorrhage or white matter lesions. 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