Mechanisms of enhanced osteoclastogenesis in girls and young women with Turner's Syndrome

Faienza, Maria Felicia; Brunetti, Giacomina; Ventura, Alessandro; Piacente, Laura; Messina, Maria Francesca; Luca, Filippo De; Ciccarelli, Maria; Oranger, Angela; Mori, Giorgio; Natale, Maria Pia; Gigante, Margherita; Ranieri, Elena; Gesualdo, Loreto; Colucci, Silvia; Grano, Maria

doi:10.1016/j.bone.2015.07.021

Cited by 28 publications

(25 citation statements)

References 47 publications

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“…VEGFA is the most important vascular endothelial growth factor, and has the strongest effect on inducing angiogenesis by binding to VEGF receptors (VEGFRs) on the surface of endothelial cells 34 . Researchers previously observed reduced bone vasculature and decreased Hif-1α and VEGF expression in the bones of OVX mice 35–37 . Our study detected that at 3 days post-injury the transcriptional levels of Hif - 1α and VEGFA in OVX rats were significantly higher than that in normal rats.…”

Section: Discussionmentioning

confidence: 98%

CD31hiEmcnhi Vessels Support New Trabecular Bone Formation at the Frontier Growth Area in the Bone Defect Repair Process

Wang

Gao

Cheng

et al. 2017

Sci Rep

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CD31hiEmcnhi vessels were a subtype of vessels in the murine skeletal system, with high levels of platelet and endothelial cell adhesion molecule-1 (PECAM-1/CD31) and endomucin (Emcn). They were reported coupling angiogenesis and osteogenesis during bone development. We investigated the distribution of these vessels in rat tibiae and their temporal and spatial distribution during the bone defect repair process to improve our understanding of the importance of these vessels. We confirmed that CD31hiEmcnhi vessels were specially distributed around the trabecular bones near metaphysis and endosteum in rat tibiae. At 3 days post bone injury, CD31hiEmcnhi vessels proliferated and were extensively distributed across the entire repair area. At 7 and 14 days post-injury, these vessels decreased but were specially distributed around the growing trabecular bones near the frontier growth area, suggesting that these vessels support new bone formation. The distribution of CD31hiEmcnhi vessels and the transcriptions of Hif-1α and VEGFA, as well as BMP2 and Osterix decreased at 7 and 14 days post-injury under osteoporotic conditions, in combination with insufficient osteogenesis. Our research is of great significance to help understand the important role of CD31hiEmcnhi vessels in supporting new trabecular bones formation during bone defect repair process.

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Section: Discussionmentioning

confidence: 98%

CD31hiEmcnhi Vessels Support New Trabecular Bone Formation at the Frontier Growth Area in the Bone Defect Repair Process

Wang

Gao

Cheng

et al. 2017

Sci Rep

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“…Bone fragility in TS patients may also be due to elevated levels of FSH in adolescence prior to HRT administration. Increased FSH levels have been associated with enhanced osteoclastogenesis, although this was also observed post HRT administration in a mechanism associated with increased RANKL expression18. These data may suggest why variable success on bone fragility and different bone density measurements are observed even after HRT administration18.…”

Section: Discussionmentioning

confidence: 99%

Body composition and bone mineral status in patients with Turner syndrome

Shi

Liu

et al. 2016

Sci Rep

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Turner syndrome (TS) is associated with decreased bone mineral density and increased fracture rate. However, the developmental trajectory of bone density or body composition in patients with TS is still unclear. The present study tested the hypothesis that different karyotypes and/or age contributes to abnormal body composition and decreased bone mineral status parameters in patients with TS. This study included 24 girls with TS, in which 13 girls exhibited X0 karyotype and 11 had mosaicism. Quantitative ultrasound (QUS) assessed the bone mineral status of the calcaneus, including bone mineral density (BMD), amplitude-dependent speed of sound (AD-SOS), broadband ultrasound attenuation (BUA) and InBody 770 assessed body composition. Pearson’s test was performed to correlate measured parameters with patient age. The body composition and bone mineral status parameters were not significantly influenced by patient karyotype. There was a correlation between patient age and AD-SOS (r = −0.61, P = 0.002) and BUA (r = 0.50, P = 0.013) but not BMD (r = −0.19, P = 0.379). In conclusion, there was no effect of karyotype on body composition or body mineral status. Bone mineral status, as evidenced by changes in AD-SOS and BUA, alters with age regardless of karyotype. The developmental trajectory demonstrated in the current study warrants further validation in a longitudinal study.

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“…Most girls with Turner syndrome, particularly those with 45,X, have high FSH at the time of expected puberty. It has been suggested that high FSH levels in untreated Turner syndrome may drive osteoclastogenesis (Faienza et al, ), although these data have not yet been confirmed.…”

Section: Endocrine Issuesmentioning

confidence: 97%

“…This can be influenced by remodeling (formation and degradation) controlled by osteoblasts and osteoclasts, respectively. In Turner syndrome, increased resorption with normal or decreased formation (imbalance of bone remodeling) may be observed (Faienza et al, ; Gravholt, Lauridsen, Brixen, et al, ). Potential mechanisms resulting in bone fragility in Turner syndrome include estrogen deficiency, FSH excess, the effect of X‐chromosome abnormality, and the comorbidities of Turner syndrome discussed separately.…”

Section: Endocrine Issuesmentioning

confidence: 99%

Recognition and management of adults with Turner syndrome: From the transition of adolescence through the senior years

Lin

Prakash

Andersen

et al. 2019

American J of Med Genetics Pt A

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Turner syndrome is recognized now as a syndrome familiar not only to pediatricians and pediatric specialists, medical geneticists, adult endocrinologists, and cardiologists, but also increasingly to primary care providers, internal medicine specialists, obstetricians, and reproductive medicine specialists. In addition, the care of women with Turner syndrome may involve social services, and various educational and neuropsychologic therapies. This article focuses on the recognition and management of Turner syndrome

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Mechanisms of enhanced osteoclastogenesis in girls and young women with Turner's Syndrome

Cited by 28 publications

References 47 publications

CD31hiEmcnhi Vessels Support New Trabecular Bone Formation at the Frontier Growth Area in the Bone Defect Repair Process

CD31hiEmcnhi Vessels Support New Trabecular Bone Formation at the Frontier Growth Area in the Bone Defect Repair Process

Body composition and bone mineral status in patients with Turner syndrome

Recognition and management of adults with Turner syndrome: From the transition of adolescence through the senior years

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