Evidence map›Paper›PMID 32039504›Full record

ReviewFASEB journal : official publication of the Federation of American Societies for Experimental Biology2020

Mechanisms underlying auditory processing deficits in Fragile X syndrome.

Elizabeth A McCullagh, Sarah E Rotschafer, Benjamin D Auerbach, Achim Klug, Leonard K Kaczmarek, Karina S Cramer, Randy J Kulesza, Khaleel A Razak, Jonathan W Lovelace, Yong Lu and 2 more

Open access · greenAbstract readReview
In one paragraph

Review in FASEB journal : official publication of the Federation of American Societies for Experimental Biology, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 48 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
48citing papers in PubMed, 2 pooled it
7.6field-weighted citation impact, top 2% 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

48 citing papers in PubMed, 2 syntheses or guidelines pooled it, 70 citations in OpenAlex.

  1. Pooled it
  2. Auditory processing in rodent models of autism: a systematic review.Journal of neurodevelopmental disorders · 2022
    Pooled it
  3. Article
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  6. Review
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  8. Article
  9. Review
  10. Article
  11. Article
  12. Atypical Retinal Ganglion Cell Function in a Mouse Model of Fragile X Syndrome.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025
    Article
  13. Hyper-extralemniscal model of Fragile X syndrome.Cerebral cortex (New York, N.Y. : 1991) · 2025
    Article
  14. Review
  15. 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

12 authors at 10 institutions in 2 countries.

Elizabeth A McCullaghDepartment of Physiology and Biophysics, University of Colorado Anschutz, Aurora, CO, USA.
Sarah E RotschaferDepartment of Neurobiology and Behavior, University of California, Irvine, CA, USA.
Benjamin D AuerbachCenter for Hearing and Deafness, Department of Communicative Disorders & Sciences, SUNY at Buffalo, Buffalo, NY, USA.
Achim KlugDepartment of Physiology and Biophysics, University of Colorado Anschutz, Aurora, CO, USA.
Leonard K KaczmarekDepartments of Pharmacology and Cellular and Molecular Physiology, Yale University, New Haven, CT, USA.
Karina S CramerDepartment of Neurobiology and Behavior, University of California, Irvine, CA, USA.
Randy J KuleszaDepartment of Anatomy, Lake Erie College of Osteopathic Medicine, Erie, PA, USA.
Khaleel A RazakDepartment of Psychology, University of California, Riverside, CA, USA.
Jonathan W LovelaceDepartment of Psychology, University of California, Riverside, CA, USA.
Yong LuDepartment of Anatomy and Neurobiology, College of Medicine, Northeast Ohio Medical University, Rootstown, OH, USA.
Ursula KochInstitute of Biology, Neurophysiology, Freie Universität Berlin, Berlin, Germany.
Yuan WangDepartment of Biomedical Sciences, Program in Neuroscience, Florida State University, Tallahassee, FL, USA.
University of California, Riverside · USUniversity of Colorado Anschutz Medical Campus · USFlorida State University · USFreie Universität Berlin · DELake Erie College of Osteopathic Medicine · USMercer University · USNortheast Ohio Medical University · USUniversity of California, Irvine · USWomen & Children's Hospital of Buffalo · USYale University · US

Funding

ZEBRAFISH TRANSGENIC CORE - ROCKY MOUNTAIN NEUROLOGICAL DISORDERS CORE CENTERP30NS048154 · NINDS · UNIVERSITY OF COLORADO DENVER · PI RIBERA, ANGELES BADELL · 2004 to 2020
$10.1M
Neurophysiological and acute pharmacological studies in FXS patientsU54HD082008 · NICHD · UT SOUTHWESTERN MEDICAL CENTER · PI HUBER, KIMBERLY M. · 2014 to 2019
$9.8M
EXPRESSION OF ION CHANNELS IN THE AUDITORY SYSTEMR01DC001919 · NIDCD · YALE UNIVERSITY · PI KACZMAREK, LEONARD K · 1993 to 2025
$9.7M
Cellular Regulation of Sodium-activated Potassium ChannelsR01NS102239 · NINDS · YALE UNIVERSITY · PI LEONARD K KACZMAREK · 2018 to 2026
$3.5M
Institutional Training in Otolaryngology ResearchT32DC012280 · NIDCD · UNIVERSITY OF COLORADO DENVER · PI Yuri Agrawal, Sue C. Kinnamon · 2013 to 2026
$3.4M
Glial Influences on Auditory Brainstem DevelopmentR01DC010796 · NIDCD · UNIVERSITY OF CALIFORNIA-IRVINE · PI CRAMER, KARINA S · 2011 to 2020
$3.0M
The contributions of age related changes in the sound localization pathway to central hearing lossR01DC017924 · NIDCD · UNIVERSITY OF COLORADO DENVER · PI KLUG, ACHIM, TOLLIN, DANIEL J · 2019 to 2023
$2.9M
Development and afferent regulation of auditory neuronsR01DC013074 · NIDCD · UNIVERSITY OF WASHINGTON · PI WANG, YUAN · 2014 to 2018
$1.9M
Neuronal Transdifferention in vivo: Mechanism and PotentialR01NS086839 · NINDS · UNIVERSITY OF COLORADO DENVER · PI RIBERA, ANGELES BADELL · 2014 to 2018
$1.7M
Metabotropic Glutamate Receptor-Mediated Neuromodulation in Sound Localization CircuitsR01DC016054 · NIDCD · NORTHEAST OHIO MEDICAL UNIVERSITY · PI LU, YONG · 2017 to 2021
$1.6M
Development of a Fragile X Syndrome model in the gerbil to study auditory dysfunctionR21DC017267 · NIDCD · FLORIDA STATE UNIVERSITY · PI WANG, YUAN · 2019 to 2020
$463k
The Role of Central Gain Control in Hyperacusis of Diverse OriginF32DC015160 · NIDCD · STATE UNIVERSITY OF NEW YORK AT BUFFALO · PI AUERBACH, BENJAMIN D · 2016 to 2018
$183k
HHS | NIH | National Institute on Deafness and Other Communication Disorders (NIDCD) 3T32DC012280-05S1HHS | NIH | National Institute on Deafness and Other Communication Disorders (NIDCD) F32DC015160-01A1NICHD NIH HHS U54 HD082008NIDCD NIH HHS R01 DC001919NIDCD NIH HHS R01 DC010796NIDCD NIH HHS R01 DC013074NIDCD NIH HHS R01 DC016054NIDCD NIH HHS R01 DC017924NIDCD NIH HHS R21 DC017267NIDCD NIH HHS T32 DC012280NINDS NIH HHS P30 NS048154NINDS NIH HHS R01 NS086839NINDS NIH HHS R01 NS102239
6 · The paper itself

Abstract

Autism spectrum disorders (ASD) are strongly associated with auditory hypersensitivity or hyperacusis (difficulty tolerating sounds). Fragile X syndrome (FXS), the most common monogenetic cause of ASD, has emerged as a powerful gateway for exploring underlying mechanisms of hyperacusis and auditory dysfunction in ASD. This review discusses examples of disruption of the auditory pathways in FXS at molecular, synaptic, and circuit levels in animal models as well as in FXS individuals. These examples highlight the involvement of multiple mechanisms, from aberrant synaptic development and ion channel deregulation of auditory brainstem circuits, to impaired neuronal plasticity and network hyperexcitability in the auditory cortex. Though a relatively new area of research, recent discoveries have increased interest in auditory dysfunction and mechanisms underlying hyperacusis in this disorder. This rapidly growing body of data has yielded novel research directions addressing critical questions regarding the timing and possible outcomes of human therapies for auditory dysfunction in ASD.

Indexed as

AnimalsAuditory PerceptionAutism Spectrum DisorderFragile X SyndromeHumansModels, Biologicalauditory systemautism spectrum disorderscircuit developmentFragile X syndromehyperacusissynaptic transmission

Identifiers

PMID32039504
PMCPMC7347277
OpenAlexW3005794563

What OpenQuestion holds

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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.