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Progressive subcortical gliosis (PSG) is a rare neurodegenerative dementia, which is characterized of astrocytic proliferation in the frontal and temporal lobe. Clinical symptoms of PSG include personality changes, progressive dementia, dysarthria, myoclonia, etc. Brain MRI shows T2WI hyperintense, which is related to astrocytic hyperplasia. Due to poor specification both in clinical manifestation and examination, the definite diagnosis of PSG mainly depends on the result of autopsy. The combination of neuroimage and pathology will definitly give hope to build the diagonosis of PSG while patient is living. DOI: 10.3969/j.issn.1672-6731.2017.08.012