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- W2000526757 abstract "Lung dysfunction is an important part of the pathology of the neurodegenerative disorder, Niemann–Pick C1 (NPC1). We have studied the pulmonary disease in the Npc1NIH/NIH mouse model. On histology, we find large numbers of alveolar foamy macrophages but no alveolar proteinosis. Lung weight as percent of body weight was markedly increased; using the flexiVent small animal ventilator (SCIREQ, Inc.), we find inspiratory capacity, elastance and hysterisivity to be increased while resistance was not changed. Cholesterol measurements show a doubling of lung cholesterol levels. Collagen is also increased. Treatment of Npc1−/− mice with hydroxypropyl-β-cyclodextrin (HPBCD), despite efficacious effects in brain and liver, results in little difference from age-matched controls (using a CNS-expressed transgene to extend the life expectancy of the Npc1−/− mice) for these variables." @default.
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- W2000526757 date "2011-06-01" @default.
- W2000526757 modified "2023-10-07" @default.
- W2000526757 title "Pulmonary function and pathology in hydroxypropyl-beta-cyclodextin-treated and untreated Npc1−/− mice" @default.
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- W2000526757 doi "https://doi.org/10.1016/j.ymgme.2011.03.001" @default.
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