Evidence map›Paper›PMID 35971781›Full record

ArticleAmerican journal of medical genetics. Part A2022

Expanding the phenotype of TAB2 variants and literature review.

Emily Woods, Imogen Marson, Emanuele Coci, Michael Spiller, Ajith Kumar, Angela Brady, Tessa Homfray, Richard Fisher, Peter Turnpenny, Julia Rankin and 7 more

Abstract readCase Reports
In one paragraph

Article in American journal of medical genetics. Part A, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing 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

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

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

8 citing papers in PubMed.

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  8. Expanding the phenotype of TAB2 variants and literature review.American journal of medical genetics. Part A · 2022
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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

17 authors.

Emily WoodsDepartment of Paediatrics, Sheffield Children's Hospital NHS Foundation Trust, Sheffield, UK.ORCID 0000-0003-2349-2688
Imogen MarsonMedical School, University of Sheffield, Sheffield, UK.ORCID 0000-0001-7441-831X
Emanuele CociDepartment of Pediatrics, Prignitz Hospital, Brandenburg Medical School, Prignitz, Germany.
Michael SpillerSheffield Diagnostic Genetics Service, Sheffield Children's NHS Foundation Trust, Sheffield, UK.
Ajith KumarDepartment of Clinical Genetics, Great Ormond Street Hospital, London, UK.
Angela BradyClinical Genetics Service, Northwick Park Hospital, London, UK.
Tessa HomfrayClinical Genetics Service, St George's Hospital, London, UK.
Richard FisherNorthern Genetics Service, Newcastle University Hospital NHS Trust, Newcastle, UK.
Peter TurnpennyPeninsula Clinical Genetics Service, Royal Devon and Exeter NHS Trust, Exeter, UK.
Julia RankinPeninsula Clinical Genetics Service, Royal Devon and Exeter NHS Trust, Exeter, UK.
Farah KananiSheffield Clinical Genetics Service, Sheffield Children's NHS Foundation Trust, Sheffield, UK.ORCID 0000-0003-4388-0290
Konrad PlatzerInstitute of Human Genetics, University of Leipzig Medical Center, Leipzig, Germany.
Athina VerveriDepartment of Clinical Genetics, Great Ormond Street Hospital, London, UK.
Eleftheria EmmanouilidouDepartment of Paediatrics, General Hospital of Kavala, Kavala, Greece.
Nourxan BourbounCardiology Department, AHEPA University Hospital, Aristotle University of Thessaloniki, Thessaloniki, Greece.
George GiannakoulasCardiology Department, AHEPA University Hospital, Aristotle University of Thessaloniki, Thessaloniki, Greece.ORCID 0000-0001-7491-6319
Meena BalasubramanianSheffield Clinical Genetics Service, Sheffield Children's NHS Foundation Trust, Sheffield, UK.ORCID 0000-0003-1488-3695

Funding

Medical Research Council MR/V037307/1Wellcome Trust
6 · The paper itself

Abstract

TAB2 is a gene located on chromosome 6q25.1 and plays a key role in development of the heart. Existing literature describes congenital heart disease as a common recognized phenotype of TAB2 gene variants, with evidence of a distinct syndromic phenotype also existing beyond this. Here we describe 14 newly identified individuals with nine novel, pathogenic TAB2 variants. The majority of individuals were identified through the Deciphering Developmental Disorders study through trio whole exome sequencing. Eight individuals had de novo variants, the other six individuals were found to have maternally inherited, or likely maternally inherited, variants. Five individuals from the same family were identified following cardiac disease gene panel in the proband and subsequent targeted familial gene sequencing. The clinical features of this cohort were compared to the existing literature. Common clinical features include distinctive facial features, growth abnormalities, joint hypermobility, hypotonia, and developmental delay. Newly identified features included feeding difficulties, sleep problems, visual problems, genitourinary abnormality, and other anatomical variations. Here we report 14 new individuals, including novel TAB2 variants, in order to expand the emerging syndromic clinical phenotype and provide further genotype-phenotype correlation.

Indexed as

Heart Defects, CongenitalIntellectual DisabilityAdaptor Proteins, Signal TransducingChildDevelopmental DisabilitiesExome SequencingGenetic Association StudiesHumansPhenotypeAdaptor Proteins, Signal TransducingTAB2 protein, humancongenital heart diseasedevelopmental delayfacial featuresjoint hypermobilitysyndromalTAB2

Identifiers

PMID35971781
PMCPMC9804770

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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.