Evidence map›Paper›PMID 40213817›Full record

ArticleDevelopment (Cambridge, England)2025

Targets of the transcription factor Six1 identify previously unreported candidate deafness genes.

Ramya Ranganathan, Fereshteh Sari, Scarlet Xiaoyan Wang, Alexandre Thiery, Ailin Leticia Buzzi, Rosalinda Guerra, Sally A Moody, Andrea Streit

Abstract read
In one paragraph

Article in Development (Cambridge, England), 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing 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

3 citing papers in PubMed.

  1. Article
  2. Review
  3. Sponge bHLH Gene Expression inInternational journal of molecular sciences · 2025
    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

8 authors.

Ramya RanganathanCentre for Craniofacial and Regenerative Biology, King's College London, London SE1 9RT, UK.
Fereshteh SariCentre for Craniofacial and Regenerative Biology, King's College London, London SE1 9RT, UK.
Scarlet Xiaoyan WangCentre for Craniofacial and Regenerative Biology, King's College London, London SE1 9RT, UK.
Alexandre ThieryCentre for Craniofacial and Regenerative Biology, King's College London, London SE1 9RT, UK.
Ailin Leticia BuzziCentre for Craniofacial and Regenerative Biology, King's College London, London SE1 9RT, UK.
Rosalinda GuerraCentre for Craniofacial and Regenerative Biology, King's College London, London SE1 9RT, UK.
Sally A MoodyDepartment of Anatomy & Cell Biology, George Washington University School of Medicine and Health Sciences, Washington, DC 20052, USA.
Andrea StreitCentre for Craniofacial and Regenerative Biology, King's College London, London SE1 9RT, UK.ORCID 0000-0001-7664-7917

Funding

Gene Regulation of Cranial Sensory Placode DevelopmentR01DE022065 · NIDCR · GEORGE WASHINGTON UNIVERSITY · PI MOODY, SALLY ANN · 2011 to 2015
$1.7M
Biotechnology and Biological Sciences Research Council BB/S005536/1National Institute for Health and Care Research IS-BRC-1215-20006NIDCR NIH HHS R01 DE022065NIH HHS R01 DE022065Wellcome Trust
6 · The paper itself

Abstract

Branchio-otic (BOS) and branchio-oto-renal (BOR) syndromes are autosomal dominant disorders featuring multiple birth defects including ear, renal and branchial malformations. Mutations in the homeodomain transcription factor SIX1 and its co-factor EYA1 have been identified in about 50% of individuals with BOS or BOR, while causative mutations are unknown in the other half. We hypothesise that SIX1 target genes represent new BOS and BOR candidates. Using published transcriptomic and epigenomic data from chick ear progenitors, we first identify putative Six1 targets. Next, we provide evidence that Six1 directly regulates some of these candidates: Six1 binds to their enhancers, and functional experiments in Xenopus and chick confirm that Six1 controls their expression. Finally, we show that most putative chick Six1 targets are also expressed in the human developing ear and are associated with known deafness loci. Together, our results not only characterise the molecular mechanisms that mediate Six1 function in the developing ear, but also provide new candidates for human congenital deafness.

Indexed as

DeafnessHomeodomain ProteinsXenopus ProteinsAnimalsBranchio-Oto-Renal SyndromeChick EmbryoEnhancer Elements, GeneticGene Expression Regulation, DevelopmentalHumansIntracellular Signaling Peptides and ProteinsNuclear ProteinsProtein Tyrosine PhosphatasesXenopus laevisEYA1 protein, humanHomeodomain ProteinsIntracellular Signaling Peptides and ProteinsNuclear ProteinsProtein Tyrosine PhosphatasesSIX1 protein, humanSix1 protein, XenopusXenopus ProteinsBranchio-oto-renal SyndromeDeafnessEar developmentOtic placode

Identifiers

PMID40213817
PMCPMC12045605

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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 Socratic graph.