Evidence map›Paper›PMID 15581873›Full record

ArticleDevelopmental biology2004

Otopetrin 1 is required for otolith formation in the zebrafish Danio rerio.

Inna Hughes, Brian Blasiole, David Huss, Mark E Warchol, Nigam P Rath, Belen Hurle, Elena Ignatova, J David Dickman, Ruediger Thalmann, Robert Levenson and 1 more

Open access · greenAbstract read
In one paragraph

Article in Developmental biology, 2004. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 55 papers.

0numbers the graph read from it
0cells of the map it votes in
55citing papers in PubMed
1.1field-weighted citation impact, top 26% 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

55 citing papers in PubMed, 118 citations in OpenAlex.

  1. Article
  2. Article
  3. Effects of Underwater Noise Exposure on Early Development in Zebrafish.Animals : an open access journal from MDPI · 2025
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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

11 authors at 4 institutions in 1 country.

Inna HughesDepartment of Molecular Biology and Pharmacology, Washington University Medical School, 660 South Euclid Avenue, St. Louis, MO 63110, USA.
Brian Blasiole
David Huss
Mark E Warchol
Nigam P Rath
Belen Hurle
Elena Ignatova
J David Dickman
Ruediger Thalmann
Robert Levenson
David M Ornitz
Washington University in St. Louis · USPennsylvania State University · USNational Institutes of Health · USUniversity of Missouri–St. Louis · US

Funding

Presynaptic Mechanisms of D1 Receptor Effects on Cortical FunctionP50MH068789 · NIMH · YALE UNIVERSITY · PI MCCORMICK, DAVID · 2004 to 2008
$10.3M
MECHANISMS OF SENSORY REGENERATIONR01DC006283 · NIDCD · WASHINGTON UNIVERSITY · PI WARCHOL, MARK · 2004 to 2025
$6.8M
Mechanisms regulating development of vestibular otoconiaR01DC002236 · NIDCD · WASHINGTON UNIVERSITY · PI ORNITZ, DAVID M · 1995 to 2010
$3.7M
FGF14 IN THE REGULATION OF PURKINJE NEURON EXCITABILITY AND SCA27R01NS065761 · NINDS · WASHINGTON UNIVERSITY · PI NERBONNE, JEANNE M., ORNITZ, DAVID M · 2009 to 2013
$1.9M
NIDCD NIH HHS DC006283NIDCD NIH HHS DC02236NIDCD NIH HHS R01 DC002236NIDCD NIH HHS R01 DC006283NIMH NIH HHS MH068789NIMH NIH HHS P50 MH068789NINDS NIH HHS R01 NS065761
6 · The paper itself

Abstract

Orientation with respect to gravity is essential for the survival of complex organisms. The gravity receptor is one of the phylogenetically oldest sensory systems, and special adaptations that enhance sensitivity to gravity are highly conserved. The fish inner ear contains three large extracellular biomineral particles, otoliths, which have evolved to transduce the force of gravity into neuronal signals. Mammalian ears contain thousands of small particles called otoconia that serve a similar function. Loss or displacement of these structures can be lethal for fish and is responsible for benign paroxysmal positional vertigo (BPPV) in humans. The distinct morphologies of otoconial particles and otoliths suggest divergent developmental mechanisms. Mutations in a novel gene Otopetrin 1 (Otop1), encoding multi-transmembrane domain protein, result in nonsyndromic otoconial agenesis and a severe balance disorder in mice. Here we show that the zebrafish, Danio rerio, contains a highly conserved gene, otop1, that is essential for otolith formation. Morpholino-mediated knockdown of zebrafish Otop1 leads to otolith agenesis without affecting the sensory epithelium or other structures within the inner ear. Despite lack of otoliths in early development, otolith formation partially recovers in some fish after 2 days. However, the otoliths are malformed, misplaced, lack an organic matrix, and often consist of inorganic calcite crystals. These studies demonstrate that Otop1 has an essential and conserved role in the timing of formation and the size and shape of the developing otolith.

Indexed as

AnimalsCalcium CarbonateGene Expression Regulation, DevelopmentalGravitationHumansIn Situ HybridizationIon ChannelsMembrane ProteinsMiceMicroscopy, Electron, ScanningMorphogenesisOligonucleotidesOtolithic MembraneSaccule and UtricleX-Ray DiffractionZebrafishCalcium CarbonateIon ChannelsMembrane ProteinsOligonucleotidesotop1 protein, zebrafishZebrafish ProteinsNon-programmatic

Identifiers

PMID15581873
PMCPMC2522322
OpenAlexW1984931573

What OpenQuestion holds

Textmetadata
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.