Alterations of a serum marker of collagen X in growing children with osteogenesis imperfecta.
Collagen X
Growth plate
Osteogenesis imperfecta
Journal
Bone
ISSN: 1873-2763
Titre abrégé: Bone
Pays: United States
ID NLM: 8504048
Informations de publication
Date de publication:
08 2021
08 2021
Historique:
received:
22
02
2021
revised:
12
04
2021
accepted:
25
04
2021
pubmed:
2
5
2021
medline:
10
7
2021
entrez:
1
5
2021
Statut:
ppublish
Résumé
Abnormalities in the structure and/or processing of type I collagen cause osteogenesis imperfecta and result in bone fragility, abnormal bone growth and short stature. Type I collagen is expressed in the growth plate but the mechanisms by which abnormalities in collagen I contribute to growth plate dysfunction and growth retardation are unknown. The non-collagenous domain (NC1) of type X collagen (CXM) is released from the hypertrophic zone of active growth plates and is a marker for new endochondral bone formation. Serum CXM levels are strongly correlated with the rate of growth in healthy children. We hypothesized that CXM levels in children with OI would be abnormal when compared to normally growing children. Using participants from the Brittle Bone Disease Consortium Natural History Study we analyzed the distribution of CXM over the ages of 8 months to 40 years in 187 subjects with OI (89 type I and 98 types III/IV) as well as analyzed the relationship between growth velocity and CXM levels in a subset of 100 children <16 years old with OI (44 type I and 56 types III/IV). CXM levels in both control and OI children demonstrated a similar pattern of variation by age with higher levels in early life and puberty followed by a post-pubertal drop. However, there was greater variability within the OI cohort and the relationship with growth velocity was weaker. The ratio of CXM level to growth velocity was elevated in children with type III/IV OI compared to controls. These results suggest that the relationship between hypertrophic zone function and the end point of skeletal growth is disrupted in OI.
Identifiants
pubmed: 33932621
pii: S8756-3282(21)00152-6
doi: 10.1016/j.bone.2021.115990
pmc: PMC8217291
mid: NIHMS1701186
pii:
doi:
Substances chimiques
Biomarkers
0
Collagen Type I
0
Collagen
9007-34-5
Types de publication
Journal Article
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
115990Subventions
Organisme : NIAMS NIH HHS
ID : U54 AR068069
Pays : United States
Informations de copyright
Copyright © 2021. Published by Elsevier Inc.
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