Evidence map›Paper›PMID 40083274›Full record

ArticleDisease models & mechanisms2025

CIB2 function is distinct from that of whirlin in the organization of sterocilia architecture.

Arnaud P J Giese, Andrew Parker, Sakina Rehman, Steve D M Brown, Saima Riazuddin, Craig W Vander Kooi, Michael R Bowl, Zubair M Ahmed

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Disease models & mechanisms, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

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

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. The Diverse Functions of the Calcium- and Integrin-Binding Protein Family.International journal of molecular sciences · 2025
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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

8 authors.

Arnaud P J GieseDepartment of Otorhinolaryngology - Head and Neck Surgery, University of Maryland School of Medicine, Baltimore, MD 21201, USA.
Andrew ParkerMRC Harwell Institute, Mammalian Genetics Unit, Harwell Campus, Oxfordshire OX11 0RD, UK.
Sakina RehmanDepartment of Otorhinolaryngology - Head and Neck Surgery, University of Maryland School of Medicine, Baltimore, MD 21201, USA.
Steve D M BrownMRC Harwell Institute, Mammalian Genetics Unit, Harwell Campus, Oxfordshire OX11 0RD, UK.
Saima RiazuddinDepartment of Otorhinolaryngology - Head and Neck Surgery, University of Maryland School of Medicine, Baltimore, MD 21201, USA.
Craig W Vander KooiDepartment of Biochemistry and Molecular Biology, University of Florida, Gainesville, FL 32603, USA.
Michael R BowlMRC Harwell Institute, Mammalian Genetics Unit, Harwell Campus, Oxfordshire OX11 0RD, UK.
Zubair M AhmedDepartment of Otorhinolaryngology - Head and Neck Surgery, University of Maryland School of Medicine, Baltimore, MD 21201, USA.ORCID 0000-0003-2914-4502

Funding

Usher Proteins in the Inner Ear Structure and FunctionR01DC012564 · NIDCD · UNIVERSITY OF MARYLAND BALTIMORE · PI Zubair M. Ahmed, Gregory I Frolenkov · 2013 to 2026
$7.0M
Genetics and Functional Studies of Autosomal Recessive Neurological DisordersR01NS107428 · NINDS · UNIVERSITY OF MARYLAND BALTIMORE · PI Saima Riazuddin · 2018 to 2026
$4.7M
GIPC3, multifunctional myosin adaptor in mammalian auditory hair cellsR01DC019054 · NIDCD · UNIVERSITY OF KENTUCKY · PI FROLENKOV, GREGORY I, VANDER KOOI, CRAIG · 2020 to 2024
$2.8M
Medical Research Council MC_U142684175NIDCD NIH HHS R01 DC012564NIDCD NIH HHS R01DC012564NIDCD NIH HHS R01 DC019054NINDS NIH HHS R01 NS107428University of Maryland
6 · The paper itself

Abstract

Humans and mice with mutations in genes encoding CIB2 and whirlin (WHRN) are deaf. We previously reported that CIB2 binds to WHRN and is essential for stereocilia staircase architecture of cochlear hair cells. Here, we refine the interaction domains of both proteins and show that these proteins play unique roles in stereocilia bundle formation and organization. We found that the EF2 domain of CIB2 binds to the HHD2 region of WHRN. AlphaFold2 multimer independently identified the same interacting regions and gave a thorough structural model. Next, we investigated genetic interaction between murine Cib2 and Whrn. Hearing in mice double heterozygous for functionally null alleles (Cib2KO/+;Whrnwi/+) was similar to that in age-matched wild-type mice, indicating that partial deficiency for both Cib2 and Whrn does not impair hearing. Double homozygous mutant mice (Cib2KO/KO;Whrnwi/wi) were deaf, and their cochlear stereocilia exhibited a predominant phenotype seen in single Whrnwi/wi mutants. Overexpression of WHRN in Cib2KO/KO mice did not rescue the stereocilia morphology. These data suggest that CIB2 is multifunctional, with key independent functions in the development and/or maintenance of the stereocilia staircase pattern in auditory hair cells.

Indexed as

Calcium-Binding ProteinsMembrane ProteinsStereociliaAnimalsCochleaDeafnessHair Cells, AuditoryHearingMiceMice, KnockoutModels, MolecularPhenotypeProtein BindingCalcium-Binding ProteinsMembrane ProteinsWhrn protein, mouseCIB2CochleaInner ear hair cellsStereocilia staircaseWHRN

Identifiers

PMID40083274
PMCPMC11992350

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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.