Natural helix 9 mutants of PPARγ differently affect its transcriptional activity.


Journal

Molecular metabolism
ISSN: 2212-8778
Titre abrégé: Mol Metab
Pays: Germany
ID NLM: 101605730

Informations de publication

Date de publication:
02 2019
Historique:
received: 22 10 2018
revised: 05 12 2018
accepted: 11 12 2018
pubmed: 1 1 2019
medline: 26 11 2019
entrez: 1 1 2019
Statut: ppublish

Résumé

The nuclear receptor PPARγ is the master regulator of adipocyte differentiation, distribution, and function. In addition, PPARγ induces terminal differentiation of several epithelial cell lineages, including colon epithelia. Loss-of-function mutations in PPARG result in familial partial lipodystrophy subtype 3 (FPDL3), a rare condition characterized by aberrant adipose tissue distribution and severe metabolic complications, including diabetes. Mutations in PPARG have also been reported in sporadic colorectal cancers, but the significance of these mutations is unclear. Studying these natural PPARG mutations provides valuable insights into structure-function relationships in the PPARγ protein. We functionally characterized a novel FPLD3-associated PPARγ L451P mutation in helix 9 of the ligand binding domain (LBD). Interestingly, substitution of the adjacent amino acid K450 was previously reported in a human colon carcinoma cell line. We performed a detailed side-by-side functional comparison of these two PPARγ mutants. PPARγ L451P shows multiple intermolecular defects, including impaired cofactor binding and reduced RXRα heterodimerisation and subsequent DNA binding, but not in DBD-LBD interdomain communication. The K450Q mutant displays none of these functional defects. Other colon cancer-associated PPARγ mutants displayed diverse phenotypes, ranging from complete loss of activity to wildtype activity. Amino acid changes in helix 9 can differently affect LBD integrity and function. In addition, FPLD3-associated PPARγ mutations consistently cause intra- and/or intermolecular defects; colon cancer-associated PPARγ mutations on the other hand may play a role in colon cancer onset and progression, but this is not due to their effects on the most well-studied functional characteristics of PPARγ.

Identifiants

pubmed: 30595551
pii: S2212-8778(18)31027-5
doi: 10.1016/j.molmet.2018.12.005
pmc: PMC6358588
pii:
doi:

Substances chimiques

PPAR gamma 0
PPARG protein, human 0

Types de publication

Case Reports Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

115-127

Informations de copyright

Copyright © 2018 The Authors. Published by Elsevier GmbH.. All rights reserved.

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Auteurs

Marjoleine F Broekema (MF)

Center for Molecular Medicine, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; Department of Molecular Cancer Research, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands.

Maarten P G Massink (MPG)

Center for Molecular Medicine, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; Department of Genetics, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands.

Cinzia Donato (C)

Center for Molecular Medicine, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; Department of Molecular Cancer Research, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands.

Joep de Ligt (J)

Center for Molecular Medicine, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; Department of Genetics, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands.

Joerg Schaarschmidt (J)

Bijvoet Center for Biomolecular Research, Faculty of Science, Utrecht University, Utrecht, the Netherlands.

Anouska Borgman (A)

Center for Molecular Medicine, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; Department of Molecular Cancer Research, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands.

Marieke G Schooneman (MG)

Department of Internal Medicine, Amsterdam University Medical Centers, Amsterdam, the Netherlands.

Diana Melchers (D)

PamGene International B. V., 's-Hertogenbosch, the Netherlands.

Martin N Gerding (MN)

Deventer Hospital, Deventer, the Netherlands.

René Houtman (R)

PamGene International B. V., 's-Hertogenbosch, the Netherlands.

Alexandre M J J Bonvin (AMJJ)

Bijvoet Center for Biomolecular Research, Faculty of Science, Utrecht University, Utrecht, the Netherlands.

Amit R Majithia (AR)

Division of Endocrinology, Department of Medicine, University of California San Diego, La Jolla, CA 92093, USA.

Houshang Monajemi (H)

Department of Endocrinology and Metabolism, Amsterdam University Medical Centers, Amsterdam, the Netherlands; Rijnstate Hospital, Arnhem, the Netherlands.

Gijs W van Haaften (GW)

Center for Molecular Medicine, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; Department of Genetics, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands.

Maarten R Soeters (MR)

Department of Endocrinology and Metabolism, Amsterdam University Medical Centers, Amsterdam, the Netherlands.

Eric Kalkhoven (E)

Center for Molecular Medicine, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands; Department of Molecular Cancer Research, University Medical Center Utrecht, Utrecht University, Utrecht, the Netherlands. Electronic address: e.kalkhoven@umcutrecht.nl.

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Classifications MeSH