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Record W3159564321 · doi:10.1016/j.jgg.2021.03.010

Disruption of the autism-related gene Pak1 causes stereocilia disorganization, hair cell loss, and deafness in mice

2021· article· en· W3159564321 on OpenAlexaff
Cheng Cheng, Yilin Hou, Zhonghong Zhang, Yanfei Wang, Ling Lü, Liyan Zhang, Pei Jiang, Song Gao, Qiaojun Fang, Chengwen Zhu, Junyan Gao, Xufeng Liu, Wei Xie, Zhengping Jia, Zhigang Xu, Xia Gao, Renjie Chai

Bibliographic record

VenueJournal of genetics and genomics/Journal of Genetics and Genomics · 2021
Typearticle
Languageen
FieldNeuroscience
TopicHearing, Cochlea, Tinnitus, Genetics
Canadian institutionsHospital for Sick ChildrenUniversity of Toronto
FundersNatural Science Foundation of Jiangsu ProvinceNanjing UniversityChina Postdoctoral Science FoundationChinese Academy of SciencesShenzhen Fundamental Research ProgramSoutheast UniversityNational Natural Science Foundation of China
KeywordsPAK1Stereocilia (inner ear)AutismCochleaCofilinHair cellHearing lossBiologyEzrinRadixinCell biologyKinaseGeneticsNeuroscienceActin cytoskeletonMedicineCellAudiologyCytoskeletonPsychiatry

Abstract

fetched live from OpenAlex

Several clinical studies have reported that hearing loss is correlated with autism in children. However, little is known about the underlying mechanism between hearing loss and autism. p21-activated kinases (PAKs) are a family of serine/threonine kinases that can be activated by multiple signaling molecules, particularly the Rho family of small GTPases. Previous studies have shown that Pak1 mutations are associated with autism. In the present study, we take advantage of Pak1 knockout (Pak1−/−) mice to investigate the role of PAK1 in hearing function. We find that PAK1 is highly expressed in the postnatal mouse cochlea and that PAK1 deficiency leads to hair cell (HC) apoptosis and severe hearing loss. Further investigation indicates that PAK1 deficiency downregulates the phosphorylation of cofilin and ezrin-radixin-moesin and the expression of βII-spectrin, which further decreases the HC synapse density in the basal turn of cochlea and disorganized the HC stereocilia in all three turns of cochlea in Pak1−/− mice. Overall, our work demonstrates that the autism-related gene Pak1 plays a crucial role in hearing function. As the first candidate gene linking autism and hearing loss, Pak1 may serve as a potential target for the clinical diagnosis of autism-related hearing loss.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.227
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.014
GPT teacher head0.229
Teacher spread0.215 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

Study designBench or experimental
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations40
Published2021
Admission routes1
Has abstractyes

Explore more

Same venueJournal of genetics and genomics/Journal of Genetics and GenomicsSame topicHearing, Cochlea, Tinnitus, GeneticsFrench-language works237,207