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Reversal of defective lysosomal transport in NPC disease ameliorates liver dysfunction and neurodegeneration in the npc1⁻/⁻ mouse

Proceedings of the National Academy of Sciences - PNAS, 2009-02, Vol.106 (7), p.2377-2382

2009

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A suppressor screen in Mecp2 mutant mice implicates cholesterol metabolism in Rett syndrome

Nature genetics, 2013-09, Vol.45 (9), p.1013-1020

2013

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Plant sterols and plant stanols in the management of dyslipidaemia and prevention of cardiovascular disease

Atherosclerosis, 2014-02, Vol.232 (2), p.346-360

2014

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In memoriam: John M. Dietschy Sr., MD (1932–2020)

Journal of lipid research, 2021, Vol.62, p.100015, Article 100015

2021

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Cyclodextrin overcomes the transport defect in nearly every organ of NPC1 mice leading to excretion of sequestered cholesterol as bile acid

Journal of lipid research, 2010-05, Vol.51 (5), p.933-944

2010

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Weekly Cyclodextrin Administration Normalizes Cholesterol Metabolism in Nearly Every Organ of the Niemann-Pick Type C1 Mouse and Markedly Prolongs Life

Pediatric research, 2010-10, Vol.68 (4), p.309-315

2010

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Genetic variations and treatments that affect the lifespan of the NPC1 mouse

Journal of lipid research, 2008-03, Vol.49 (3), p.663-669

2008

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Impact of loss of SOAT2 function on disease progression in the lysosomal acid lipase-deficient mouse

Steroids, 2018-02, Vol.130, p.7-14

2018

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Knockout of the Cholesterol 24-Hydroxylase Gene in Mice Reveals a Brain-specific Mechanism of Cholesterol Turnover

The Journal of biological chemistry, 2003-06, Vol.278 (25), p.22980-22988

2003

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Unesterified cholesterol accumulation in late endosomes/lysosomes causes neurodegeneration and is prevented by driving cholesterol export from this compartment

The Journal of neuroscience, 2011-06, Vol.31 (25), p.9404-9413

2011

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Suppression of brain cholesterol synthesis in male Mecp2-deficient mice is age dependent and not accompanied by a concurrent change in the rate of fatty acid synthesis

Brain research, 2017-01, Vol.1654 (Pt A), p.77-84

2017

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Control of Cholesterol Turnover in the Mouse

The Journal of biological chemistry, 2002-02, Vol.277 (6), p.3801-3804

2002

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Quantitation of the rates of hepatic and intestinal cholesterol synthesis in lysosomal acid lipase-deficient mice before and during treatment with ezetimibe

Biochemical pharmacology, 2017-07, Vol.135, p.116-125

2017

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Molecular markers of brain cholesterol homeostasis are unchanged despite a smaller brain mass in a mouse model of cholesteryl ester storage disease

Lipids, 2022-01, Vol.57 (1), p.3-16

2022

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Ontogenesis and Modulation of Intestinal Unesterified Cholesterol Sequestration in a Mouse Model of Niemann–Pick C1 Disease

Digestive diseases and sciences, 2020-01, Vol.65 (1), p.158-167

2020

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Niemann-Pick C1-deficient mice lacking sterol O-acyltransferase 2 have less hepatic cholesterol entrapment and improved liver function

American journal of physiology: Gastrointestinal and liver physiology, 2018-10, Vol.315 (4), p.G454-G463

2018

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Liver X Receptor Activation Enhances Cholesterol Loss from the Brain, Decreases Neuroinflammation, and Increases Survival of the NPC1 Mouse

The Journal of neuroscience, 2007-12, Vol.27 (52), p.14470-14480

2007

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Deletion of sterol O-acyltransferase 2 (SOAT2) function in mice deficient in lysosomal acid lipase (LAL) dramatically reduces esterified cholesterol sequestration in the small intestine and liver

Biochemical and biophysical research communications, 2014-11, Vol.454 (1), p.162-166

2014

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Quantitative role of LAL, NPC2, and NPC1 in lysosomal cholesterol processing defined by genetic and pharmacological manipulations

Journal of lipid research, 2011-04, Vol.52 (4), p.688-698

2011

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Measurement of Rates of Cholesterol and Fatty Acid Synthesis In Vivo Using Tritiated Water

Methods in molecular biology (Clifton, N.J.), 2017-01, Vol.1583, p.241

2017

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Hepatic entrapment of esterified cholesterol drives continual expansion of whole body sterol pool in lysosomal acid lipase-deficient mice

American journal of physiology: Gastrointestinal and liver physiology, 2014-10, Vol.307 (8), p.G836-G847

2014

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Delineation of metabolic responses of Npc1−/−nih mice lacking the cholesterol-esterifying enzyme SOAT2 to acute treatment with 2-hydroxypropyl-β-cyclodextrin

Steroids, 2020-12, Vol.164, p.108725-108725, Article 108725

2020

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Ezetimibe markedly attenuates hepatic cholesterol accumulation and improves liver function in the lysosomal acid lipase-deficient mouse, a model for cholesteryl ester storage disease

Biochemical and biophysical research communications, 2014-01, Vol.443 (3), p.1073-1077

2014

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Inhibiting intestinal NPC1L1 activity prevents diet-induced increase in biliary cholesterol in Golden Syrian hamsters

American journal of physiology: Gastrointestinal and liver physiology, 2008-10, Vol.295 (4), p.G813-G822

2008

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ABCA1 plays no role in the centripetal movement of cholesterol from peripheral tissues to the liver and intestine in the mouse

Journal of lipid research, 2009-07, Vol.50 (7), p.1316-1329

2009

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Reversal of defective lysosomal transport in NPC disease ameliorates liver dysfunction and neurodegeneration in the npc1⁻/⁻ mouse

A suppressor screen in Mecp2 mutant mice implicates cholesterol metabolism in Rett syndrome

Plant sterols and plant stanols in the management of dyslipidaemia and prevention of cardiovascular disease

In memoriam: John M. Dietschy Sr., MD (1932–2020)

Cyclodextrin overcomes the transport defect in nearly every organ of NPC1 mice leading to excretion of sequestered cholesterol as bile acid

Weekly Cyclodextrin Administration Normalizes Cholesterol Metabolism in Nearly Every Organ of the Niemann-Pick Type C1 Mouse and Markedly Prolongs Life

Genetic variations and treatments that affect the lifespan of the NPC1 mouse

Impact of loss of SOAT2 function on disease progression in the lysosomal acid lipase-deficient mouse

Knockout of the Cholesterol 24-Hydroxylase Gene in Mice Reveals a Brain-specific Mechanism of Cholesterol Turnover

Unesterified cholesterol accumulation in late endosomes/lysosomes causes neurodegeneration and is prevented by driving cholesterol export from this compartment

Suppression of brain cholesterol synthesis in male Mecp2-deficient mice is age dependent and not accompanied by a concurrent change in the rate of fatty acid synthesis

Control of Cholesterol Turnover in the Mouse

Quantitation of the rates of hepatic and intestinal cholesterol synthesis in lysosomal acid lipase-deficient mice before and during treatment with ezetimibe

Molecular markers of brain cholesterol homeostasis are unchanged despite a smaller brain mass in a mouse model of cholesteryl ester storage disease

Ontogenesis and Modulation of Intestinal Unesterified Cholesterol Sequestration in a Mouse Model of Niemann–Pick C1 Disease

Niemann-Pick C1-deficient mice lacking sterol O-acyltransferase 2 have less hepatic cholesterol entrapment and improved liver function

Liver X Receptor Activation Enhances Cholesterol Loss from the Brain, Decreases Neuroinflammation, and Increases Survival of the NPC1 Mouse

Deletion of sterol O-acyltransferase 2 (SOAT2) function in mice deficient in lysosomal acid lipase (LAL) dramatically reduces esterified cholesterol sequestration in the small intestine and liver

Quantitative role of LAL, NPC2, and NPC1 in lysosomal cholesterol processing defined by genetic and pharmacological manipulations

Measurement of Rates of Cholesterol and Fatty Acid Synthesis In Vivo Using Tritiated Water

Hepatic entrapment of esterified cholesterol drives continual expansion of whole body sterol pool in lysosomal acid lipase-deficient mice

Delineation of metabolic responses of Npc1−/−nih mice lacking the cholesterol-esterifying enzyme SOAT2 to acute treatment with 2-hydroxypropyl-β-cyclodextrin

Ezetimibe markedly attenuates hepatic cholesterol accumulation and improves liver function in the lysosomal acid lipase-deficient mouse, a model for cholesteryl ester storage disease

Inhibiting intestinal NPC1L1 activity prevents diet-induced increase in biliary cholesterol in Golden Syrian hamsters

ABCA1 plays no role in the centripetal movement of cholesterol from peripheral tissues to the liver and intestine in the mouse

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