Evidence map›Paper›PMID 39062269›Full record

ArticleChildren (Basel, Switzerland)2024

Sclerostin and Wnt Signaling in Idiopathic Juvenile Osteoporosis Using High-Resolution Confocal Microscopy for Three-Dimensional Analyses.

Renata C Pereira, Kathleen J Noche, Barbara Gales, Zhangying Chen, Isidro B Salusky, Lauren V Albrecht

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Article in Children (Basel, Switzerland), 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
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1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

6 authors.

Renata C PereiraDepartment of Pediatrics, David School of Medicine, University of California Los Angeles, Los Angeles, CA 90024, USA.
Kathleen J NocheDepartment of Pediatrics, David School of Medicine, University of California Los Angeles, Los Angeles, CA 90024, USA.
Barbara GalesDepartment of Pediatrics, David School of Medicine, University of California Los Angeles, Los Angeles, CA 90024, USA.
Zhangying ChenDepartment of Pharmaceutical Sciences, School of Pharmacy and Pharmaceutical Sciences, University of California Irvine, Irvine, CA 92697, USA.
Isidro B SaluskyDepartment of Pediatrics, David School of Medicine, University of California Los Angeles, Los Angeles, CA 90024, USA.
Lauren V AlbrechtDepartment of Pharmaceutical Sciences, School of Pharmacy and Pharmaceutical Sciences, University of California Irvine, Irvine, CA 92697, USA.ORCID 0000-0002-3147-8010

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundIdiopathic juvenile osteoporosis (IJO) is a rare condition characterized by low bone mass that can increase the risk of fractures in children. Treatment options for these patients are limited as the molecular mechanisms of disease initiation and progression are incompletely understood. Sclerostin inhibits canonical Wnt signaling, which is important for the bone formation activity of osteoblasts, and elevated sclerostin has been implicated in adult osteoporosis.

objectiveTo evaluate the role of sclerostin in IJO, high-resolution confocal microscopy analyses were performed on bone biopsies collected from 13 pediatric patients.

methodsBone biopsies were stained with sclerostin, and β-catenin antibodies showed elevated expression across osteocytes and increased sclerostin-positive osteocytes in 8 of the 13 total IJO patients (62%).

resultsSkeletal sclerostin was associated with static and dynamic histomorphometric parameters. Further, colocalization analyses showed that bone sclerostin colocalized with phosphorylated β-catenin, a hallmark of Wnt signaling that indicates Wnt inhibition. In contrast, sclerostin-positive osteocytes were not colocalized with an "active" unphosphorylated form of β-catenin.

conclusionsThese results support a model that altered levels of sclerostin and Wnt signaling activity occur in IJO patients.

Indexed as

bone biopsychildrenimmunofluorescenceosteoporosissclerostinWnt signaling

Identifiers

PMID39062269
PMCPMC11276078

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