Development of the Bone Phenotype and microRNA Profile in Adults With Low-Density Lipoprotein Receptor-Related Protein 5-High Bone Mass (LRP5-HBM) Disease.

HIGH BONE MASS HR‐pQCT LRP5 RARE MONOGENETIC BONE DISEASE microRNA

Journal

JBMR plus
ISSN: 2473-4039
Titre abrégé: JBMR Plus
Pays: England
ID NLM: 101707013

Informations de publication

Date de publication:
Sep 2021
Historique:
received: 27 01 2021
revised: 07 07 2021
accepted: 19 07 2021
entrez: 17 9 2021
pubmed: 18 9 2021
medline: 18 9 2021
Statut: epublish

Résumé

Pathogenic variants in the Wnt-pathway co-receptor low-density lipoprotein (LDL) receptor-related protein 5 (LRP5) cause high bone mass (LRP5-HBM) due to insensitivity to the endogenous antagonist of Wnt-signaling. Although indicating incessant progression of BMD and biomarkers reflecting bone formation, this has not been confirmed in individuals with LRP5-HBM. We investigated how the LRP5-HBM bone phenotype changes with age in adults and is associated with quantitative changes of bone turnover markers and bone-related microRNAs (miRNAs) in the circulation. Whole body, lumbar spine, total hip, and femoral neck areal BMD (aBMD) and radial and tibial bone microarchitecture and geometry were assessed using DXA and HR-pQCT scans of 15 individuals with LRP5-HBM

Identifiants

pubmed: 34532618
doi: 10.1002/jbm4.10534
pii: JBM410534
pmc: PMC8441296
doi:

Types de publication

Journal Article

Langues

eng

Pagination

e10534

Subventions

Organisme : Medical Research Council
ID : MR/P020941/1
Pays : United Kingdom

Informations de copyright

© 2021 The Authors. JBMR Plus published by Wiley Periodicals LLC. on behalf of American Society for Bone and Mineral Research.

Déclaration de conflit d'intérêts

MH and MW report personal fees from TAmiRNA GmbH, outside the submitted work. RE reports grants from Amgen, grants and personal fees from IDS, grants from Alexion, grants and personal fees from Roche, personal fees from GSK Nutrition, personal fees from Mereo, personal fees from Sandoz, grants and personal fees from Nittobo, personal fees from AbbVie, personal fees from Samsung, personal fees from Haoma Medica, personal fees from Elsevier, personal fees from CL Bio, personal fees from FNIH, personal fees from Viking, personal fees from UCSF, personal fees from Biocon, from Lyramid, outside the submitted work.

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Auteurs

Jens-Jacob Lindegaard Lauterlein (JL)

Department of Endocrinology and Metabolism Odense University Hospital Odense Denmark.
Department of Clinical Research University of Southern Denmark Odense Denmark.

Fatma Gossiel (F)

Department of Oncology and Metabolism University of Sheffield Sheffield UK.

Moritz Weigl (M)

TAmiRNA GmbH Vienna Austria.

Richard Eastell (R)

Department of Oncology and Metabolism University of Sheffield Sheffield UK.

Pernille Hermann (P)

Department of Endocrinology and Metabolism Odense University Hospital Odense Denmark.
Department of Clinical Research University of Southern Denmark Odense Denmark.

Jens Bollerslev (J)

Department of Endocrinology Rikshospitalet Oslo Norway.
Faculty of Medicine University of Oslo Oslo Norway.

Morten Frost (M)

Department of Endocrinology and Metabolism Odense University Hospital Odense Denmark.
Department of Clinical Research University of Southern Denmark Odense Denmark.
Steno Diabetes Centre Odense Odense University Hospital Odense Denmark.

Classifications MeSH