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PMID: 24531328 Published · ppublish English Journal Article Research Support, N.I.H., Extramural

Exome-wide association study identifies a TM6SF2 variant that confers susceptibility to nonalcoholic fatty liver disease.

Nature genetics ·Vol. 46 ·No. 4 ·2014-04-00 ·Pages 352-6

Kozlitina J, Smagris E, Stender S, Nordestgaard BG, Zhou HH, Tybjærg-Hansen A, Vogt TF, Hobbs HH, Cohen JC

Abstract

Nonalcoholic fatty liver disease (NAFLD) is the most common form of liver disease. To elucidate the molecular basis of NAFLD, we performed an exome-wide association study of liver fat content. Three variants were associated with higher liver fat levels at the exome-wide significance level of 3.6 × 10(-7): two in PNPLA3, an established locus for NAFLD, and one (encoding p.Glu167Lys) in TM6SF2, a gene of unknown function. The TM6SF2 variant encoding p.Glu167Lys was also associated with higher circulating levels of alanine transaminase, a marker of liver injury, and with lower levels of low-density lipoprotein-cholesterol (LDL-C), triglycerides and alkaline phosphatase in 3 independent populations (n > 80,000). When recombinant protein was expressed in cultured hepatocytes, 50% less Glu167Lys TM6SF2 protein was produced relative to wild-type TM6SF2. Adeno-associated virus-mediated short hairpin RNA knockdown of Tm6sf2 in mice increased liver triglyceride content by threefold and decreased very-low-density lipoprotein (VLDL) secretion by 50%. Taken together, these data indicate that TM6SF2 activity is required for normal VLDL secretion and that impaired TM6SF2 function causally contributes to NAFLD.

MeSH Terms
Adipose Tissue/metabolism Alanine Transaminase/blood Amino Acid Sequence Animals Base Sequence Chromatography, Liquid Dependovirus Exome/genetics Fatty Liver/genetics Gene Knockdown Techniques Genetic Association Studies Genetic Predisposition to Disease/genetics Hepatocytes Humans Lipoproteins, VLDL/metabolism Liver/metabolism Membrane Proteins/genetics Mice Molecular Sequence Data Mutation, Missense/genetics Non-alcoholic Fatty Liver Disease Real-Time Polymerase Chain Reaction Recombinant Proteins/genetics,metabolism Sequence Alignment Sequence Analysis, DNA Triglycerides/metabolism
Chemicals
Lipoproteins, VLDL Membrane Proteins Recombinant Proteins TM6SF2 protein, human Triglycerides Alanine Transaminase
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Kozlitina Julia
1] McDermott Center for Human Growth and Development, University of Texas Southwestern Medical Center, Dallas, Texas, USA. [2].
Smagris Eriks
1] McDermott Center for Human Growth and Development, University of Texas Southwestern Medical Center, Dallas, Texas, USA. [2].
Stender Stefan
Department of Clinical Biochemistry, Rigshospitalet, Copenhagen University Hospital and Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark.
Nordestgaard Børge G
1] Copenhagen General Population Study, Herlev Hospital, Copenhagen University Hospital and Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark. [2] Department of Clinical Biochemistry, Herlev Hospital, Copenhagen University Hospital and Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark. [3] Copenhagen City Heart Study, Frederiksberg Hospital, Copenhagen University Hospital and Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark.
Zhou Heather H
Merck Research Laboratories, Kenilworth, New Jersey, USA.
Tybjærg-Hansen Anne
1] Department of Clinical Biochemistry, Rigshospitalet, Copenhagen University Hospital and Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark. [2] Copenhagen General Population Study, Herlev Hospital, Copenhagen University Hospital and Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark. [3] Copenhagen City Heart Study, Frederiksberg Hospital, Copenhagen University Hospital and Faculty of Health and Medical Sciences, University of Copenhagen, Copenhagen, Denmark.
Vogt Thomas F
Merck Research Laboratories, Kenilworth, New Jersey, USA.
Hobbs Helen H
1] McDermott Center for Human Growth and Development, University of Texas Southwestern Medical Center, Dallas, Texas, USA. [2] Howard Hughes Medical Institute, University of Texas Southwestern Medical Center, Dallas, Texas, USA.
Cohen Jonathan C
McDermott Center for Human Growth and Development, University of Texas Southwestern Medical Center, Dallas, Texas, USA.
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Article Info
Journal
Nature genetics
Abbr.
Nat Genet
ISSN
1546-1718
Published
2014-04-00
Epub
2014-00-16
Pages
352-6
Language
English
Region
United States
NLM ID
9216904
PMCID
PMC3969786
Subset
IM
Grants
NHLBI NIH HHS · 1HL092550 · United States
NHLBI NIH HHS · HL20948, · United States
NHLBI NIH HHS · RL1 HL092550 · United States
NCATS NIH HHS · UL1TR001105 · United States
NIDDK NIH HHS · DK090066 · United States
NIDDK NIH HHS · R01 DK090066 · United States
NHLBI NIH HHS · P01 HL020948 · United States
NCATS NIH HHS · UL1 TR001105 · United States
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