Evidence map›Paper›PMID 40360853›Full record

ArticleJournal of the Association for Research in Otolaryngology : JARO2025

Haploinsufficiency of Whrn Contributes to Progressive Sensorineural Hearing Loss in C57BL6 Mice.

Han-Gyu Bae, Sean Kashiwagura, Andrew Jung, Elizabeth Gould, Jun Hee Kim

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Article in Journal of the Association for Research in Otolaryngology : JARO, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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1 · What the graph read from it

What it found

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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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3 · Its place in the literature

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4 · The record

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5 · Who and what money

Authors and funding

5 authors.

Han-Gyu BaeDepartment of Otolaryngology Head and Neck Surgery, Kresge Hearing Research Institute, University of Michigan, Ann Arbor, MI, USA.
Sean KashiwaguraDepartment of Cellular and Integrative Physiology, University of Texas Health Science Center, San Antonio, USA.
Andrew JungDepartment of Cellular and Integrative Physiology, University of Texas Health Science Center, San Antonio, USA.
Elizabeth GouldDepartment of Cellular and Integrative Physiology, University of Texas Health Science Center, San Antonio, USA.
Jun Hee KimDepartment of Otolaryngology Head and Neck Surgery, Kresge Hearing Research Institute, University of Michigan, Ann Arbor, MI, USA. kimjunh@umich.edu.ORCID http://orcid.org/0000-0003-0207-8410

Funding

Role of Adaptive Myelination in Auditory Brain PlasticityR01DC018797 · NIDCD · UNIVERSITY OF TEXAS HLTH SCIENCE CENTER · PI Jun Hee Kim · 2021 to 2026
$2.9M
NIDCD NIH HHS R01 DC018797
6 · The paper itself

Abstract

purposeWhrn, encoding whirlin, is one of the genes highly relevant to Usher syndrome (USH) that has been known as an autosomal recessive genetic disorder that is characterized with sensorineural hearing loss with retinitis pigmentosa. Although recent studies on the other USH genes, PDZD7 and Ush1 g, showed a possibility of haploinsufficiency effect, the potential contribution of heterozygous Whrn loss to hearing loss remains unclear.

methodsTo investigate the effect of Whrn haploinsufficiency, we conducted a longitudinal study assessing auditory function in heterozygous Whrn mutant (Whrn

resultsHearing threshold was significantly increased with aging in Whrn

conclusionThese results provide evidence for a haploinsufficiency effect of Whrn on auditory function and highlight its potential role in progressive sensorineural hearing loss.

Indexed as

HaploinsufficiencyHearing Loss, SensorineuralAnimalsEvoked Potentials, Auditory, Brain StemFemaleMaleMiceMice, Inbred C57BLAgingHearing lossSexUsher syndrome type 2Whirlin

Identifiers

PMID40360853
PMCPMC12133618

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