Home LiteratureArticle Details
PMID: 18512226 Published · ppublish English Case Reports Journal Article Review

A further example of a distinctive autosomal recessive syndrome comprising neonatal diabetes mellitus, intestinal atresias and gall bladder agenesis.

American journal of medical genetics. Part A ·Vol. 146A ·No. 13 ·2008-07-01 ·Pages 1713-7

Chappell L, Gorman S, Campbell F, Ellard S, Rice G, Dobbie A, Crow Y

Abstract

We report a patient born to consanguineous parents as a further example of a recently described phenotype comprising neonatal diabetes, intestinal atresias and gall bladder agenesis. Other reports have described cases with overlapping patterns including malrotation, biliary atresia and pancreatic hypoplasia (e.g. as described by Martínez-Frías). We propose that these cases may represent variations of the same syndrome. It is likely that this disorder is inherited as an autosomal recessive trait. Our case is the first to have neonatal diabetes without a demonstrable structural pancreatic abnormality, showing that a deficit in pancreatic function is involved. We sequenced genes with a recognized role in monogenic forms of diabetes, including KCNJ11, ABCC8, GCK, IPF1, HNF1beta, NeuroD1 and TCF7L2, as well as a novel candidate gene, HNF6, known to be involved in hepatobiliary and pancreatic development, but did not identify mutations.

MeSH Terms
Abnormalities, Multiple/genetics Consanguinity DNA Mutational Analysis Diabetes Mellitus/congenital,genetics Female Gallbladder/abnormalities Genes, Recessive Hepatocyte Nuclear Factor 6/genetics Humans Infant Infant, Newborn Intestinal Atresia/complications,genetics Phenotype Syndrome
Chemicals
Hepatocyte Nuclear Factor 6
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Chappell Louise
School of Medicine, University of Leeds, Leeds, UK.
Gorman Shaun
Campbell Fiona
Ellard Sian
Rice Gillian
Dobbie Angus
Crow Yanick
Article Info
Journal
American journal of medical genetics. Part A
Abbr.
Am J Med Genet A
ISSN
1552-4833
Published
2008-07-01
Pages
1713-7
Language
English
Region
United States
NLM ID
101235741
Subset
IM
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: [email protected]