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PMID: 15281001 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Triple genetic variation in the HNF-4alpha gene is associated with early-onset type 2 diabetes mellitus in a philippino family.

Metabolism: clinical and experimental ·Vol. 53 ·No. 8 ·2004-08-00 ·Pages 959-63

Gragnoli C, Menzinger Von Preussenthal G, Habener JF

Abstract

Maturity-onset diabetes of the young-type 1 (MODY1) is a form of monogenic type 2 diabetes mellitus (T2DM) with long-term complications due to mutations in the HNF-4alpha gene. The HNF-4alpha gene is involved in hepatic differentiation and expression of genes regulating glucose transport, glycolysis, and lipid metabolism. The abnormal glucose-stimulated insulin secretion in MODY1 subjects may be due to reduced glucose transport and glycolysis. To date, 14 mutations in the HNF-4alpha gene have been identified as a cause of either MODY1 or late-onset type 2 diabetes. So far, no screening has been performed in subjects from the Philippines. We recruited a Philippino family with autosomal dominant early-onset type 2 diabetes and screened the proband for mutations in the genes for HNF-1alpha, GCK, HNF-4alpha, IPF-1, HNF-6, and NGN3. We identified a new missense mutation in exon 5 (V199I) of the HNF-4alpha gene and 2 new single-nucleotide substitutions in intron 4, IVS4-nt4 (G --> A) and IVS4-nt20 (C --> T), all cosegregating with diabetes in the 3 affected available siblings. These variations were not present in 100 normal healthy subjects. Bioinformatic analysis suggests that these variations in the whole, and overall the IVS4-nt4 variation located at splicing site, may affect the splicing potential of intron 4. We have biochemically and clinically characterized the Philippine-1 family. We suggest that the V199I missense mutation located in the ligand binding/dimerization domain of HNF-4alpha contributes to type 2 diabetes in the Philippine-1 family. The intron variations may contribute susceptibility to diabetes.

MeSH Terms
Adult Age of Onset Aged DNA/genetics DNA-Binding Proteins/genetics Diabetes Mellitus, Type 2/blood,genetics Exons/genetics Female Hepatocyte Nuclear Factor 4 Humans Islets of Langerhans/metabolism Male Middle Aged Mutation, Missense/genetics,physiology Pedigree Phenotype Philippines Phosphoproteins/genetics Transcription Factors/genetics,metabolism
Chemicals
DNA-Binding Proteins HNF4A protein, human Hepatocyte Nuclear Factor 4 Phosphoproteins Transcription Factors DNA
Authors & Affiliations
3 authors, click to expand affiliations / ORCID
Gragnoli Claudia
Laboratory of Molecular Endocrinology, Massachusetts General Hospital, Harvard Medical School, Boston 02114, USA.
Menzinger Von Preussenthal Guido
Habener Joel F
Article Info
Journal
Metabolism: clinical and experimental
Abbr.
Metabolism
ISSN
0026-0495
Published
2004-08-00
Pages
959-63
Language
English
Region
United States
NLM ID
0375267
Subset
IM
Grants
NIDDK NIH HHS · DK-55365 · United States
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