Evidence map›Paper›PMID 39713379›Full record

ArticlebioRxiv : the preprint server for biology2024

High resolution kinematic approach for quantifying impaired mobility of dystrophic zebrafish larvae.

Jeffrey J Widrick, Matthias R Lambert, Felipe de Souza Leite, Youngsook Lucy Jung, Junseok Park, James R Conner, Eunjung Alice Lee, Alan H Beggs, Louis M Kunkel

Abstract readPreprint
In one paragraph

Article in bioRxiv : the preprint server for biology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
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

1 citing paper in PubMed.

  1. Article
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

9 authors.

Jeffrey J WidrickDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.ORCID 0000-0002-5597-1141
Matthias R LambertDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.
Felipe de Souza LeiteDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.
Youngsook Lucy JungDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.
Junseok ParkDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.
James R ConnerDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.
Eunjung Alice LeeDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.
Alan H BeggsDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.
Louis M KunkelDivision of Genetics and Genomics, Dept. of Pediatrics, Boston Children's Hospital, Boston, MA.

Funding

Genetic Analysis and Manipulation Core (GAEC)P50HD105351 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI Hisashi Umemori · 2021 to 2026
$9.4M
Modulation of Jagged1/Pitpna in DMD as a means of therapyR01AR064300 · NIAMS · BOSTON CHILDREN'S HOSPITAL · PI KUNKEL, LOUIS M · 2014 to 2023
$3.9M
Unveiling the molecular mechanisms in TPM3-related myopathy and therapiesK99AR080197 · NIAMS · BOSTON CHILDREN'S HOSPITAL · PI LAMBERT, MATTHIAS · 2022 to 2023
$212k
NIAMS NIH HHS K99 AR080197NIAMS NIH HHS R01 AR064300NICHD NIH HHS P50 HD105351
6 · The paper itself

Abstract

Dystrophin-deficient zebrafish larvae are a small, genetically tractable vertebrate model of Duchenne muscular dystrophy well suited for early stage therapeutic development. However, current approaches for evaluating their impaired mobility, a physiologically relevant therapeutic target, are characterized by low resolution and high variability. To address this, we used high speed videography and deep learning-based markerless motion capture to develop linked-segment models of larval escape response (ER) swimming. Kinematic models provided repeatable, high precision estimates of larval ER performance. Effect sizes for ER peak instantaneous acceleration and speed, final displacement, and ER distance were 2 to 3.5 standard deviations less for dystrophin-deficient mutants vs. wild-types. Further analysis revealed that mutants swam slower because of a reduction in their tail stroke frequency with little change in tail stroke amplitude. Kinematic variables were highly predictive of the dystrophic phenotype with ≤ 3% of larvae misclassified by random forest and support vector machine models. Tail kinematics also performed as well as

Indexed as

DeepLabCutDuchenne muscular dystrophyescape responsepose estimationsapjesapje-likeswimming

Identifiers

PMID39713379
PMCPMC11661059

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.