Evidence map›Paper›PMID 31051468›Full record

ArticleEndocrinology, diabetes & metabolism case reports2019

Familial GATA6 mutation causing variably expressed diabetes mellitus and cardiac and renal abnormalities.

Yang Timothy Du, Lynette Moore, Nicola K Poplawski, Sunita M C De Sousa

Abstract read
In one paragraph

Article in Endocrinology, diabetes & metabolism case reports, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

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Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

5 citing papers in PubMed.

  1. Neonatal diabetes mellitus around the world: Update 2024.Journal of diabetes investigation · 2024
    Review
  2. Review
  3. Article
  4. Clinical characteristics and genetic analysis of a child with specific type of diabetes mellitus caused by missense mutation ofZhejiang da xue xue bao. Yi xue ban = Journal of Zhejiang University. Medical sciences · 2023
    Article
  5. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

4 authors.

Yang Timothy DuEndocrine and Metabolic Unit, Royal Adelaide Hospital.
Lynette MooreSchool of Medicine, University of Adelaide.
Nicola K PoplawskiAdult Genetics Unit, Royal Adelaide Hospital.
Sunita M C De SousaEndocrine and Metabolic Unit, Royal Adelaide Hospital.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

A 26-year-old man presented with a combination of permanent neonatal diabetes due to pancreatic aplasia, complex congenital heart disease, central hypogonadism and growth hormone deficiency, structural renal abnormalities with proteinuria, umbilical hernia, neurocognitive impairment and dysmorphic features. His older brother had diabetes mellitus due to pancreatic hypoplasia, complex congenital heart disease, hypospadias and umbilical hernia. Their father had an atrial septal defect, umbilical hernia and diabetes mellitus diagnosed incidentally in adulthood on employment screening. The proband's paternal grandmother had a congenital heart defect. Genetic testing of the proband revealed a novel heterozygous missense variant (Chr18:g.19761441T>C, c.1330T>C, p.Cys444Arg) in exon 4 of GATA6, which is class 5 (pathogenic) using American College of Medical Genetics and Genomics guidelines and is likely to account for his multisystem disorder. The same variant was detected in his brother and father, but not his paternal grandmother. This novel variant of GATA6 likely occurred de novo in the father with autosomal dominant inheritance in the proband and his brother. The case is exceptional as very few families with monogenic diabetes due to GATA6 mutations have been reported to date and we describe a new link between GATA6 and renal pathology. Learning points: Monogenic diabetes should be suspected in patients presenting with syndromic features, multisystem congenital disease, neonatal-onset diabetes and/or a suggestive family history. Recognition and identification of genetic diabetes may improve patient understanding and empowerment and allow for better tailored management. Identification of a genetic disorder may have important implications for family planning.

Indexed as

2019AdultAustraliaCardiologyCardiovascular endocrinologyCognitive problemsDevelopmental endocrinologyDiabetesDNA sequencingFacial plethoraFacies - abnormalGenetic analysisGeneticsGenetics and mutationGHGonadotropinsGrowth hormone deficiencyGrowth hormone deficiency (adult)Haematoxylin and eosin stainingHeartHistopathologyHypogonadismImmunostainingInsulinKidneyMaleMayMolecular genetic analysisNeonatal diabetesNew disease or syndrome: presentations/diagnosis/managementPaediatric endocrinologyPaediatricsPancreasPituitaryProteinuriaRenal biopsyUmbilical herniaUrologyWhite

Identifiers

PMID31051468
PMCPMC6499914

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.