Evidence map›Paper›PMID 40085271›Full record

ArticleCalcified tissue international2025

Loss of the Ubiquitin-Associated Domain of sqstm1/p62 in Zebrafish Causes a Phenotype Resembling Paget's Disease of Bone.

Yentl Huybrechts, Raphaël De Ridder, Dylan Bergen, Björn De Samber, Eveline Boudin, Francesca Tonelli, Dries Knapen, Lucia Vergauwen, Dorien Schepers, Evelien Van Dijck and 13 more

Abstract read
PubMed Publisher
In one paragraph

Article in Calcified tissue international, 2025. 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

23 authors.

Yentl Huybrechts *Center of Medical Genetics, University of Antwerp and University Hospital Antwerp, Antwerp, Belgium.
Raphaël De Ridder *Center of Medical Genetics, University of Antwerp and University Hospital Antwerp, Antwerp, Belgium.
Dylan BergenHammond Lab, School of Physiology, Pharmacology, and Neuroscience, University of Bristol, Bristol, UK.
Björn De SamberImec-Vision Lab, Department of Physics, University of Antwerp, Antwerp, Belgium.
Eveline BoudinCenter of Medical Genetics, University of Antwerp and University Hospital Antwerp, Antwerp, Belgium.
Francesca TonelliDepartment of Molecular Medicine, Biochemistry Unit, University of Pavia, Pavia, Italy.
Dries KnapenZebrafishlab, Veterinary Physiology and Biochemistry, Department of Veterinary Sciences, University of Antwerp, Antwerp, Belgium.
Lucia VergauwenZebrafishlab, Veterinary Physiology and Biochemistry, Department of Veterinary Sciences, University of Antwerp, Antwerp, Belgium.
Dorien SchepersCenter of Medical Genetics, University of Antwerp and University Hospital Antwerp, Antwerp, Belgium.
Evelien Van DijckCenter of Medical Genetics, University of Antwerp and University Hospital Antwerp, Antwerp, Belgium.
Qiao TongHammond Lab, School of Physiology, Pharmacology, and Neuroscience, University of Bristol, Bristol, UK.
Anja VerhulstLaboratory of Pathophysiology, University of Antwerp, Antwerp, Belgium.
Jan De BeenhouwerImec-Vision Lab, Department of Physics, University of Antwerp, Antwerp, Belgium.
Jan SijbersImec-Vision Lab, Department of Physics, University of Antwerp, Antwerp, Belgium.
Chrissy HammondHammond Lab, School of Physiology, Pharmacology, and Neuroscience, University of Bristol, Bristol, UK.
Antonella ForlinoDepartment of Molecular Medicine, Biochemistry Unit, University of Pavia, Pavia, Italy.
Geert MortierLaboratory for Skeletal Dysplasia Research, Department of Human Genetics, KU Leuven and University Hospital Leuven, Louvain, Belgium.
Paul CouckeCenter of Medical Genetics Ghent, Department of Biomolecular Medicine, Ghent University, Ghent, Belgium.
P Eckhard WittenEvolutionary Developmental Biology Group, Department of Biology, Ghent University, Ghent, Belgium.
Ronald Young KwonDepartment of Orthopaedics and Sports Medicine, School of Medicine, University of Washington, Seattle, WA, USA.
Andy WillaertCenter of Medical Genetics Ghent, Department of Biomolecular Medicine, Ghent University, Ghent, Belgium.
Gretl Hendrickx *Center of Medical Genetics, University of Antwerp and University Hospital Antwerp, Antwerp, Belgium.
Wim Van Hul *Center of Medical Genetics, University of Antwerp and University Hospital Antwerp, Antwerp, Belgium. wim.vanhul@uantwerpen.be.ORCID http://orcid.org/0000-0002-5065-7858

Funding

FWO 12R5623NNational institute of health AR074417Universiteit Antwerpen FFB190208University of Ghent BOF.GOA.2021.0004Versus Arthritis 21937Versus Arthritis 22044
6 · The paper itself

Abstract

The ubiquitin-binding protein p62, encoded by Sequestosome 1 (SQSTM1), is an essential molecular adaptor for selective autophagy. Heterozygous mutations deleting or disrupting the ubiquitin-associated (UBA) domain of p62 have been reported as the major genetic cause for Paget's disease of bone (PDB), the second most common skeletal disease, characterized by hyperactive osteoclasts and focal increases of bone turnover. In this study, we aimed to determine the impact of a similar sqstm1/p62 mutation on the skeleton of zebrafish. We successfully established a sqstm1

Indexed as

Osteitis DeformansSequestosome-1 ProteinZebrafish ProteinsAnimalsAutophagyBone and BonesDisease Models, AnimalMutationOsteoclastsPhenotypeZebrafishSequestosome-1 ProteinZebrafish ProteinsGenetic animal modelPaget’s disease of bonesqstm1UBAZebrafish

Identifiers

PMID40085271

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.