Comparison of bone microstructure and strength in the distal radius and tibia between the different types of primary hypertrophic osteoarthropathy: an HR-pQCT study.
DKK1
HR-pQCT
PHO
PHOAR1
PHOAR2
Journal
Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA
ISSN: 1433-2965
Titre abrégé: Osteoporos Int
Pays: England
ID NLM: 9100105
Informations de publication
Date de publication:
Aug 2023
Aug 2023
Historique:
received:
20
09
2022
accepted:
01
05
2023
medline:
31
7
2023
pubmed:
19
5
2023
entrez:
18
5
2023
Statut:
ppublish
Résumé
Primary hypertrophic osteoarthropathy (PHO) is a hereditary bone disease that is grouped into PHO autosomal recessive 1 (PHOAR1) and PHO autosomal recessive 2 (PHOAR2) due to different causative genes. Data comparing bone microstructure between the two subtypes are scarce. This is the first study to find that PHOAR1 patients had inferior bone microstructure compared with PHOAR2 patients. The primary goal of this study was to assess bone microarchitecture and strength in PHOAR1 and PHOAR2 patients and to compare them with age- and sex-matched healthy controls (HCs). The secondary goal was to assess the differences between PHOAR1 and PHOAR2 patients. Twenty-seven male Chinese PHO patients (PHOAR1 = 7; PHOAR2 = 20) were recruited from Peking Union Medical College Hospital. The areal bone mineral density (aBMD) was assessed by dual-energy X-ray absorptiometry (DXA). Peripheral bone microarchitecture at the distal radius and tibia were evaluated by high-resolution peripheral quantitative computed tomography (HR-pQCT). Biochemical markers of PGE2, bone turnover, and Dickkopf-1 (DKK1) were investigated. Compared with HCs, PHOAR1 and PHOAR2 patients had distinctively larger bone geometry, substantially lower vBMD at the radius and tibia, and compromised cortical microstructure at the radius. For trabecular bone, PHOAR1 and PHOAR2 patients showed different changes at the tibia. PHOAR1 patients had significant deficits in the trabecular compartment, resulting in lower estimated bone strength. Conversely, PHOAR2 patients showed a higher trabecular number, narrower trabecular separation, and lower trabecular network inhomogeneity than HCs, translating into preserved or slightly high estimated bone strength. PHOAR1 patients had inferior bone microstructure and strength compared with PHOAR2 patients and HCs. Additionally, this study was the first to find differences in the bone microstructure between PHOAR1 and PHOAR2 patients.
Identifiants
pubmed: 37202541
doi: 10.1007/s00198-023-06784-w
pii: 10.1007/s00198-023-06784-w
pmc: PMC10382400
doi:
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
1453-1464Subventions
Organisme : National Key R&D Program of China
ID : 2018YFA0800801
Organisme : Key Research and Development Projects of Shaanxi Province
ID : 2021YFC2501700
Organisme : CAMS Innovation Fund for Medical Sciences
ID : CIFMS-2021-I2M-1-002
Organisme : CAMS Innovation Fund for Medical Sciences
ID : 2020-I2M-C&T-B-016
Organisme : National Natural Science Foundation of China
ID : 81970757
Organisme : National Natural Science Foundation of China
ID : 81900798
Organisme : Institute of Chinese Materia Medica, China Academy of Chinese Medical Sciences and Peking Union Medical College
ID : ZC201904197
Organisme : grant from Peking Union Medical College Hospital
ID : 2017PT32020
Organisme : grant from Peking Union Medical College Hospital
ID : 2018PT32001
Informations de copyright
© 2023. The Author(s).
Références
Am J Hum Genet. 2012 Jan 13;90(1):125-32
pubmed: 22197487
Nat Genet. 2008 Jun;40(6):789-93
pubmed: 18500342
J Bone Miner Res. 2020 Nov;35(11):2159-2170
pubmed: 32564403
J Bone Miner Res. 2014 Sep;29(9):2118-29
pubmed: 24644043
Arthritis Rheum. 1993 Oct;36(10):1444-50
pubmed: 8216404
J Clin Invest. 1996 Sep 1;98(5):1142-9
pubmed: 8787677
J Bone Miner Res. 2017 Aug;32(8):1659-1666
pubmed: 28425581
Osteoporos Int. 2020 Jan;31(1):153-164
pubmed: 31646353
J Musculoskelet Neuronal Interact. 2007 Oct-Dec;7(4):372-81
pubmed: 18094515
Am J Pathol. 2010 Feb;176(2):721-32
pubmed: 20042673
Bone. 2000 Oct;27(4):487-94
pubmed: 11033443
Bone. 2007 Sep;41(3):331-9
pubmed: 17613296
J Bone Miner Res. 2015 Jan;30(1):176-83
pubmed: 25042721
J Bone Miner Res. 2022 Mar;37(3):484-493
pubmed: 34894003
J Bone Miner Res. 2021 Aug;36(8):1459-1468
pubmed: 33852188
Am J Med. 2007 Nov;120(11):940-1
pubmed: 17976417
Calcif Tissue Int. 2018 Jan;102(1):105-116
pubmed: 29105022
J Bone Miner Res. 2010 Oct;25(10):2229-38
pubmed: 20499344
Osteoporos Int. 2020 Sep;31(9):1607-1627
pubmed: 32458029
Bone. 2014 Apr;61:109-15
pubmed: 24412702
J Bone Miner Res. 2013 Apr;28(4):794-806
pubmed: 23090909
J Lipid Mediat Cell Signal. 1995 Oct;12(2-3):313-9
pubmed: 8777575
Bone. 2018 Jan;106:96-102
pubmed: 28963081
J Bone Miner Res. 1997 Feb;12(2):276-82
pubmed: 9041061
Bone. 2012 Jun;50(6):1304-10
pubmed: 22445540