Evidence map›Paper›PMID 31217345›Full record

ArticleJCI insight2019

TRIOBP-5 sculpts stereocilia rootlets and stiffens supporting cells enabling hearing.

Tatsuya Katsuno, Inna A Belyantseva, Alexander X Cartagena-Rivera, Keisuke Ohta, Shawn M Crump, Ronald S Petralia, Kazuya Ono, Risa Tona, Ayesha Imtiaz, Atteeq Rehman and 15 more

Open access · goldAbstract read
In one paragraph

Article in JCI insight, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.

0numbers the graph read from it
0cells of the map it votes in
30citing papers in PubMed
3.5field-weighted citation impact, top 7% of its field
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

30 citing papers in PubMed, 47 citations in OpenAlex.

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  11. Combined AAV-mediated gene replacement therapy improves auditory function in a mouse model of human DFNB42 deafness.Molecular therapy : the journal of the American Society of Gene Therapy · 2023
    Article
  12. Review
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  14. SUMOylation targeting mitophagy in cardiovascular diseases.Journal of molecular medicine (Berlin, Germany) · 2022
    Review
  15. Unbalanced bidirectional radial stiffness gradients within the organ of Corti promoted by TRIOBP.Proceedings of the National Academy of Sciences of the United States of America · 2022
    Article
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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

25 authors at 6 institutions in 2 countries.

Tatsuya KatsunoDepartment of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.
Inna A BelyantsevaLaboratory of Molecular Genetics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Alexander X Cartagena-RiveraSection on Auditory Mechanics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Keisuke OhtaAdvanced Imaging Research Center, Kurume University School of Medicine, Kurume, Japan.
Shawn M CrumpDepartment of Physiology, University of Kentucky, Lexington, Kentucky, USA.
Ronald S PetraliaAdvanced Imaging Core, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Kazuya OnoDepartment of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.
Risa TonaDepartment of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.
Ayesha ImtiazLaboratory of Molecular Genetics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Atteeq RehmanLaboratory of Molecular Genetics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Hiroshi KiyonariLaboratory for Animal Resources and Genetic Engineering, Riken Center for Biosystems Dynamics Research, Kobe, Japan.
Mari KanekoLaboratory for Animal Resources and Genetic Engineering, Riken Center for Biosystems Dynamics Research, Kobe, Japan.
Ya-Xian WangAdvanced Imaging Core, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Takaya AbeLaboratory for Animal Resources and Genetic Engineering, Riken Center for Biosystems Dynamics Research, Kobe, Japan.
Makoto IkeyaDepartment of Clinical Application, Center for iPS Cell Research and Application, Kyoto University, Kyoto, Japan.
Cristina Fenollar-FerrerLaboratory of Molecular Genetics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Gavin P RiordanLaboratory of Molecular Genetics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Elisabeth A WilsonLaboratory of Molecular Genetics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Tracy S FitzgeraldMouse Auditory Testing Core Facility, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Kohei SegawaDepartment of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.
Koichi OmoriDepartment of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.
Juichi ItoDepartment of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.
Gregory I FrolenkovDepartment of Physiology, University of Kentucky, Lexington, Kentucky, USA.
Thomas B FriedmanLaboratory of Molecular Genetics, National Institute on Deafness and Other Communication Disorders, NIH, Bethesda, Maryland, USA.
Shin-Ichiro KitajiriDepartment of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.
National Institute on Deafness and Other Communication Disorders · USKyoto University · JPRIKEN Center for Biosystems Dynamics Research · JPUniversity of Kentucky · USKurume University · JPShinshu University · JP

Funding

Identification of Genes Causing Syndromic And Nonsyndromic Hearing ImpairmentZIADC000039 · NIDCD · NATIONAL INSTITUTE ON DEAFNESS AND OTHER COMMUNICATION DISORDERS · PI FRIEDMAN, THOMAS · 2009 to 2025
$48.7M
Structure, Function and Pharmacology of Neurotransmitter Reuptake SystemsZIAMH002946 · NIMH · NATIONAL INSTITUTE OF MENTAL HEALTH · PI AMARA, SUSAN · 2013 to 2025
$26.6M
Myosin XVA and Other Genes Essential for Stereocilia MorphogenesisZIADC000048 · NIDCD · NATIONAL INSTITUTE ON DEAFNESS AND OTHER COMMUNICATION DISORDERS · PI FRIEDMAN, THOMAS · 2009 to 2025
$23.6M
Advanced Imaging CoreZICDC000081 · NIDCD · NATIONAL INSTITUTE ON DEAFNESS AND OTHER COMMUNICATION DISORDERS · PI WINKLER, DENNIS · 2012 to 2025
$17.4M
Mouse Auditory Testing Core FacilityZICDC000080 · NIDCD · NATIONAL INSTITUTE ON DEAFNESS AND OTHER COMMUNICATION DISORDERS · PI FITZGERALD, TRACY · 2012 to 2025
$8.0M
Identification of Genes Causing Syndromic And Nonsyndromic Hearing ImpairmentZ01DC000039 · NIDCD · NATIONAL INSTITUTE ON DEAFNESS AND OTHER COMMUNICATION DISORDERS · PI FRIEDMAN, THOMAS BAER · 1997 to 2008
$7.2M
Repair of mechano-electrical transduction in mammalian auditory hair cellsR01DC014658 · NIDCD · UNIVERSITY OF KENTUCKY · PI FROLENKOV, GREGORY I · 2015 to 2019
$2.0M
SPEECH AND HEARING SCIENCEST32DC000039 · NIDCD · CUNY GRADUATE SCH AND UNIV CTR · PI SCHWARTZ, RICHARD G · 1992 to 2002
$429k
Intramural NIH HHS Z01 DC000039Intramural NIH HHS ZIA MH002946Intramural NIH HHS ZIC DC000081NIDCD NIH HHS R01 DC014658NIDCD NIH HHS T32 DC000039
6 · The paper itself

Abstract

TRIOBP remodels the cytoskeleton by forming unusually dense F-actin bundles and is implicated in human cancer, schizophrenia, and deafness. Mutations ablating human and mouse TRIOBP-4 and TRIOBP-5 isoforms are associated with profound deafness, as inner ear mechanosensory hair cells degenerate after stereocilia rootlets fail to develop. However, the mechanisms regulating formation of stereocilia rootlets by each TRIOBP isoform remain unknown. Using 3 new Triobp mouse models, we report that TRIOBP-5 is essential for thickening bundles of F-actin in rootlets, establishing their mature dimensions and for stiffening supporting cells of the auditory sensory epithelium. The coiled-coil domains of this isoform are required for reinforcement and maintenance of stereocilia rootlets. A loss of TRIOBP-5 in mouse results in dysmorphic rootlets that are abnormally thin in the cuticular plate but have increased widths and lengths within stereocilia cores, and causes progressive deafness recapitulating the human phenotype. Our study extends the current understanding of TRIOBP isoform-specific functions necessary for life-long hearing, with implications for insight into other TRIOBPopathies.

Indexed as

ActinsAnimalsDeafnessHearingMiceMice, KnockoutMicrofilament ProteinsProtein IsoformsStereociliaActinsMicrofilament ProteinsProtein IsoformsTRIOBP protein, mouseCytoskeletonNeuroscience

Identifiers

PMID31217345
PMCPMC6629139
OpenAlexW2950588021

What OpenQuestion holds

Textmetadata
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 OpenQuestion graph.