Evidence map›Paper›PMID 29037661›Full record

ArticleBehavioural brain research2018

Antisense oligonucleotide therapy rescues disruptions in organization of exploratory movements associated with Usher syndrome type 1C in mice.

Tia N Donaldson, Kelsey T Jennings, Lucia A Cherep, Adam M McNeela, Frederic F Depreux, Francine M Jodelka, Michelle L Hastings, Douglas G Wallace

Abstract read
In one paragraph

Article in Behavioural brain research, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.

0numbers the graph read from it
0cells of the map it votes in
16citing papers in PubMed
0.9field-weighted citation impact, top 29% 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

16 citing papers in PubMed, 26 citations in OpenAlex.

  1. Article
  2. Advancing precision ear medicine: leveraging animal models for disease insights and therapeutic innovations.Mammalian genome : official journal of the International Mammalian Genome Society · 2025
    Review
  3. Article
  4. Review
  5. Article
  6. Article
  7. Article
  8. Alternative splicing in shaping the molecular landscape of the cochlea.Frontiers in cell and developmental biology · 2023
    Review
  9. Article
  10. Article
  11. Review
  12. Article
  13. Article
  14. Review
  15. Review
  16. Antisense Oligonucleotides for the Treatment of Inner Ear Dysfunction.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2019
    Review
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

8 authors at 2 institutions in 1 country.

Tia N DonaldsonNorthern Illinois University, Department of Psychology, DeKalb, IL 60115, United States.
Kelsey T JenningsNorthern Illinois University, Department of Psychology, DeKalb, IL 60115, United States.
Lucia A CherepNorthern Illinois University, Department of Psychology, DeKalb, IL 60115, United States.
Adam M McNeelaNorthern Illinois University, Department of Psychology, DeKalb, IL 60115, United States.
Frederic F DepreuxThe Chicago Medical School, Rosalind Franklin University of Medicine and Science, Department of Cell Biology and Anatomy, North Chicago, IL 60064, United States.
Francine M JodelkaThe Chicago Medical School, Rosalind Franklin University of Medicine and Science, Department of Cell Biology and Anatomy, North Chicago, IL 60064, United States.
Michelle L HastingsThe Chicago Medical School, Rosalind Franklin University of Medicine and Science, Department of Cell Biology and Anatomy, North Chicago, IL 60064, United States.
Douglas G WallaceNorthern Illinois University, Department of Psychology, DeKalb, IL 60115, United States. Electronic address: dwallace@niu.edu.
Northern Illinois University · USRosalind Franklin University of Medicine and Science · US

Funding

Correction of Hearing and Vestibular Defects in a Mouse Model for DeafnessR01DC012596 · NIDCD · ROSALIND FRANKLIN UNIV OF MEDICINE & SCI · PI HASTINGS, MICHELLE L · 2013 to 2016
$1.3M
NIDCD NIH HHS R01 DC012596
6 · The paper itself

Abstract

Usher syndrome, Type 1C (USH1C) is an autosomal recessive inherited disorder in which a mutation in the gene encoding harmonin is associated with multi-sensory deficits (i.e., auditory, vestibular, and visual). USH1C (Usher) mice, engineered with a human USH1C mutation, exhibit these multi-sensory deficits by circling behavior and lack of response to sound. Administration of an antisense oligonucleotide (ASO) therapeutic that corrects expression of the mutated USH1C gene, has been shown to increase harmonin levels, reduce circling behavior, and improve vestibular and auditory function. The current study evaluates the organization of exploratory movements to assess spatial organization in Usher mice and determine the efficacy of ASO therapy in attenuating any such deficits. Usher and heterozygous mice received the therapeutic ASO, ASO-29, or a control, non-specific ASO treatment at postnatal day five. Organization of exploratory movements was assessed under dark and light conditions at two and six-months of age. Disruptions in exploratory movement organization observed in control-treated Usher mice were consistent with impaired use of self-movement and environmental cues. In general, ASO-29 treatment rescued organization of exploratory movements at two and six-month testing points. These observations are consistent with ASO-29 rescuing processing of multiple sources of information and demonstrate the potential of ASO therapies to ameliorate topographical disorientation associated with other genetic disorders.

Indexed as

AnimalsBehavior, AnimalCarrier ProteinsCell Cycle ProteinsCytoskeletal ProteinsExploratory BehaviorMaleMiceMovementOligonucleotides, AntisenseUsher SyndromesCarrier ProteinsCell Cycle ProteinsCytoskeletal ProteinsOligonucleotides, AntisenseUsh1c protein, mouseAntisense oligonucleotidesDead reckoningDeafnessHarmoninHome basePath integrationPre-mRNA splicingSpatial orientationUSH1CUsher syndromeVestibular systemVisual system

Identifiers

PMID29037661
PMCPMC5683898
OpenAlexW2762013291

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

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.