Evidence map›Paper›PMID 39417583›Full record

ArticleDevelopment (Cambridge, England)2024

Planar cell polarity zebrafish models of congenital scoliosis reveal underlying defects in notochord morphogenesis.

Mingqin Wang, Sen Zhao, Chenjun Shi, Marie-Claude Guyot, Meijiang Liao, Josephine T Tauer, Bettina M Willie, Nikita Cobetto, Carl-Éric Aubin, Elke Küster-Schöck and 4 more

Erratum issuedAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Development (Cambridge, England), 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing 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

3 citing papers in PubMed.

  1. Caveolae mechanics in cellular functions and disease.Nature reviews. Molecular cell biology · 2026
    Review
  2. Article
  3. Review
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

14 authors.

Mingqin WangAzrieli Research Center of CHU Sainte Justine, University of Montreal, Montreal H3T 1C5, QC, Canada.ORCID 0009-0001-9445-389X
Sen ZhaoThe Department of Orthopedic Surgery, Key Laboratory of Big Data for Spinal Deformities, Beijing Key Laboratory for Genetic Research of Skeletal Deformity, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China.ORCID 0000-0001-9418-4231
Chenjun ShiDepartment of Biomedical Engineering, Wayne State University, Detroit, MI 48201, USA.
Marie-Claude GuyotAzrieli Research Center of CHU Sainte Justine, University of Montreal, Montreal H3T 1C5, QC, Canada.
Meijiang LiaoThe CHUM Research Center, University of Montréal, Montréal H2X 0A9, Canada.
Josephine T TauerFaculty of Dental Medicine and Oral Health Sciences, McGill University, Shriners Hospital for Children-Canada, Montreal H4A 0A9, QC, Canada.ORCID 0000-0002-6079-4653
Bettina M WillieFaculty of Dental Medicine and Oral Health Sciences, McGill University, Shriners Hospital for Children-Canada, Montreal H4A 0A9, QC, Canada.ORCID 0000-0003-2907-3580
Nikita CobettoDepartment Mechanical Engineering, Polytechnique Montreal, Montreal H3T 1J4, QC, Canada.
Carl-Éric AubinAzrieli Research Center of CHU Sainte Justine, University of Montreal, Montreal H3T 1C5, QC, Canada.ORCID 0000-0003-2374-008X
Elke Küster-SchöckAzrieli Research Center of CHU Sainte Justine, University of Montreal, Montreal H3T 1C5, QC, Canada.ORCID 0000-0002-9487-6948
Pierre DrapeauDepartment of Neurosciences, University of Montreal, Montreal H3C 3J7, QC, Canada.ORCID 0000-0001-9063-0142
Jitao ZhangDepartment of Biomedical Engineering, Wayne State University, Detroit, MI 48201, USA.
Nan WuThe Department of Orthopedic Surgery, Key Laboratory of Big Data for Spinal Deformities, Beijing Key Laboratory for Genetic Research of Skeletal Deformity, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China.
Zoha KibarAzrieli Research Center of CHU Sainte Justine, University of Montreal, Montreal H3T 1C5, QC, Canada.ORCID 0009-0004-6554-0339

Funding

Optically mapping tissue biomechanics during neural tube closureK25HD097288 · NICHD · WAYNE STATE UNIVERSITY · PI ZHANG, JITAO · 2019 to 2023
$562k
Canada Foundation for InnovationCentre de recherche Azrieli du CHU Sainte-JustineFonds de Recherche du Québec - SantéInstitut TransMedTechNatural Sciences and Engineering Research Council of CanadaNICHD NIH HHS K25 HD097288NIH HHS K25HD097288Richard Barber Interdisciplinary Research ProgramShriners Hospitals for Children
6 · The paper itself

Abstract

Congenital scoliosis (CS) is a type of vertebral malformation for which the etiology remains elusive. The notochord is pivotal for vertebrae development, but its role in CS is still understudied. Here, we generated a zebrafish knockout of ptk7a, a planar cell polarity (PCP) gene that is essential for convergence and extension (C&E) of the notochord, and detected congenital scoliosis-like vertebral malformations (CVMs). Maternal zygotic ptk7a mutants displayed severe C&E defects of the notochord. Excessive apoptosis occurred in the malformed notochord, causing a significantly reduced number of vacuolated cells, and compromising the mechanical properties of the notochord. The latter manifested as a less-stiff extracellular matrix along with a significant reduction in the number of the caveolae and severely loosened intercellular junctions in the vacuolated region. These defects led to focal kinks, abnormal mineralization, and CVMs exclusively at the anterior spine. Loss of function of another PCP gene, vangl2, also revealed excessive apoptosis in the notochord associated with CVMs. This study suggests a new model for CS pathogenesis that is associated with defects in notochord C&E and highlights an essential role of PCP signaling in vertebrae development.

Indexed as

ApoptosisCell PolarityMorphogenesisNotochordScoliosisZebrafishZebrafish ProteinsAnimalsDisease Models, AnimalExtracellular MatrixIntercellular JunctionsMembrane ProteinsNerve Tissue ProteinsReceptor Protein-Tyrosine KinasesSpineMembrane ProteinsNerve Tissue ProteinsReceptor Protein-Tyrosine Kinasesvangl2 protein, zebrafishZebrafish ProteinsCongenital scoliosisNotochordPlanar cell polarityZebrafish model

Identifiers

PMID39417583
PMCPMC11698040

What Socratic 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 Socratic graph.