Evidence map›Paper›PMID 32970669›Full record

ArticlePLoS genetics2020

Aldh inhibitor restores auditory function in a mouse model of human deafness.

Guang-Jie Zhu, Sihao Gong, Deng-Bin Ma, Tao Tao, Wei-Qi He, Linqing Zhang, Fang Wang, Xiao-Yun Qian, Han Zhou, Chi Fan and 11 more

Open access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in PLoS genetics, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.

0numbers the graph read from it
0cells of the map it votes in
10citing papers in PubMed
0.9field-weighted citation impact, top 24% 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

10 citing papers in PubMed, 23 citations in OpenAlex.

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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

21 authors at 5 institutions in 2 countries.

Guang-Jie ZhuDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Sihao GongDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Deng-Bin MaDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Tao TaoDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.ORCID 0000-0003-4066-8171
Wei-Qi HeDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.ORCID 0000-0003-3137-8420
Linqing ZhangDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Fang WangDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.ORCID 0000-0003-2280-3923
Xiao-Yun QianDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Han ZhouDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Chi FanDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Pei WangDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.ORCID 0000-0002-0538-7641
Xin ChenDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Wei ZhaoDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Jie SunDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.ORCID 0000-0002-6855-9076
Huaqun ChenCollege of Life Science, Nanjing Normal University, Nanjing, China.ORCID 0000-0002-3860-5887
Ye WangNanjing MuCyte Biotechnology Co., Ltd., Nanjing, China.
Xiang GaoDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Jian ZuoDepartment of Biomedical Sciences, School of Medicine, Creighton University, United States of America.ORCID 0000-0002-1448-4507
Min-Sheng ZhuDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.ORCID 0000-0003-3256-6861
Xia GaoDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.
Guoqiang WanDepartment of Otorhinolaryngology, Provincial Key Discipline of the affiliated Drum Tower Hospital of Nanjing University and Model Animal Research Center, MOE Key Laboratory of Model Animal for Disease Studies, School of Medicine, Nanjing University, Nanjing, China.ORCID 0000-0003-3841-9301
Model Animal Research Center · CNNanjing Brain Hospital · CNCreighton University · USNanjing Normal University · CNSoochow University · CN

Funding

GENETIC ANALYSIS OF MAMMALIAN HEARING SENSITIVITYR01DC006471 · NIDCD · ST. JUDE CHILDREN'S RESEARCH HOSPITAL · PI ZUO, JIAN · 2004 to 2013
$3.5M
NIDCD NIH HHS R01 DC006471
6 · The paper itself

Abstract

Genetic hearing loss is a common health problem with no effective therapy currently available. DFNA15, caused by mutations of the transcription factor POU4F3, is one of the most common forms of autosomal dominant non-syndromic deafness. In this study, we established a novel mouse model of the human DFNA15 deafness, with a Pou4f3 gene mutation (Pou4f3Δ) identical to that found in a familial case of DFNA15. The Pou4f3(Δ/+) mice suffered progressive deafness in a similar manner to the DFNA15 patients. Hair cells in the Pou4f3(Δ/+) cochlea displayed significant stereociliary and mitochondrial pathologies, with apparent loss of outer hair cells. Progression of hearing and outer hair cell loss of the Pou4f3(Δ/+) mice was significantly modified by other genetic and environmental factors. Using Pou4f3(-/+) heterozygous knockout mice, we also showed that DFNA15 is likely caused by haploinsufficiency of the Pou4f3 gene. Importantly, inhibition of retinoic acid signaling by the aldehyde dehydrogenase (Aldh) and retinoic acid receptor inhibitors promoted Pou4f3 expression in the cochlear tissue and suppressed the progression of hearing loss in the mutant mice. These data demonstrate Pou4f3 haploinsufficiency as the main underlying cause of human DFNA15 deafness and highlight the therapeutic potential of Aldh inhibitors for treatment of progressive hearing loss.

Indexed as

Aldehyde DehydrogenaseAnimalsBenzaldehydesDisease Models, AnimalEnzyme InhibitorsHair Cells, AuditoryHaploinsufficiencyHearing LossHomeodomain ProteinsHumansMice, Inbred C57BLMice, Inbred StrainsMice, KnockoutNoisepara-AminobenzoatesQuinolines4-(5,6-dihydro-5,5-dimethyl-8-(quinolin-3-yl)naphthalen-2-carboxamido)benzoic acid4-(diethylamino)benzaldehydeAldehyde DehydrogenaseBenzaldehydesEnzyme InhibitorsHomeodomain Proteinspara-AminobenzoatesPOU4F3 protein, humanPou4f3 protein, mouseQuinolinesTranscription Factor Brn-3CTretinoin

Identifiers

PMID32970669
PMCPMC7553308
OpenAlexW3089321509

What OpenQuestion holds

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LicenceCC BY
Read underepoch 390

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.