A cross-sectional and longitudinal analysis of trabecular bone score in adults with turner syndrome

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Abstract

Context: Turner syndrome (TS) is associated with short stature, gonadal failure, and fractures. Spinal trabecular bone score (TBS) is a novel bone imaging modality that has not been evaluated in TS. Objective: To evaluate TBS in TS and its association with bone mineral density (BMD), prevalent fracture, and risk factors. Design and Setting: Longitudinal study of TS from a single tertiary hospital between 2006 and 2017. Patients or Other Participants: Fifty-eight subjects with TS aged 20 to 49 years who underwent dualenergy X-ray absorptiometry (DXA). Main Outcome Measures: TBS, DXA parameters, and prevalent fractures were investigated. Results: Normal, partially degraded, and degraded TBSs were observed in 39 (67%), 15 (26%), and four (7%) subjects, respectively. High rates of prescribed estrogen replacement therapy (ERT) with stable TBS and BMD were observed during follow-up. TBS was positively correlated with spine and femoral neck (FN) BMD and Z-scores (all P<0.05) and negatively correlated with age (20.004 per year; P = 0.014) and delay in ERT initiation inwomenwith primary amenorrhea (20.010 per year; P<0.001). Fractureswere present in 17 (31%) subjects. LowTBS had a significantly higher area under the receiver operator curve for predicting prevalent fracture than low bone mass at either the spine or FN (P < 0.05). Subjects with no history of fracture were more likely to have a normal TBS (P = 0.023). Conclusions: BMD and TBS can be preserved with early initiation and continued use of ERT. TBS may provide additional fracture risk prediction to standard DXA parameters in TS and needs to be validated in larger prospective studies.

Original languageEnglish
Pages (from-to)3792-3800
Number of pages9
JournalJournal of Clinical Endocrinology and Metablism
Volume103
Issue number10
DOIs
Publication statusPublished - 1 Oct 2018

Cite this

@article{5df998d7e6b2486bb481a1858068795d,
title = "A cross-sectional and longitudinal analysis of trabecular bone score in adults with turner syndrome",
abstract = "Context: Turner syndrome (TS) is associated with short stature, gonadal failure, and fractures. Spinal trabecular bone score (TBS) is a novel bone imaging modality that has not been evaluated in TS. Objective: To evaluate TBS in TS and its association with bone mineral density (BMD), prevalent fracture, and risk factors. Design and Setting: Longitudinal study of TS from a single tertiary hospital between 2006 and 2017. Patients or Other Participants: Fifty-eight subjects with TS aged 20 to 49 years who underwent dualenergy X-ray absorptiometry (DXA). Main Outcome Measures: TBS, DXA parameters, and prevalent fractures were investigated. Results: Normal, partially degraded, and degraded TBSs were observed in 39 (67{\%}), 15 (26{\%}), and four (7{\%}) subjects, respectively. High rates of prescribed estrogen replacement therapy (ERT) with stable TBS and BMD were observed during follow-up. TBS was positively correlated with spine and femoral neck (FN) BMD and Z-scores (all P<0.05) and negatively correlated with age (20.004 per year; P = 0.014) and delay in ERT initiation inwomenwith primary amenorrhea (20.010 per year; P<0.001). Fractureswere present in 17 (31{\%}) subjects. LowTBS had a significantly higher area under the receiver operator curve for predicting prevalent fracture than low bone mass at either the spine or FN (P < 0.05). Subjects with no history of fracture were more likely to have a normal TBS (P = 0.023). Conclusions: BMD and TBS can be preserved with early initiation and continued use of ERT. TBS may provide additional fracture risk prediction to standard DXA parameters in TS and needs to be validated in larger prospective studies.",
author = "Nguyen, {Hanh Huu} and Phillip Wong and Strauss, {Boyd J} and Ebeling, {Peter R} and Frances Milat and Amanda Vincent",
year = "2018",
month = "10",
day = "1",
doi = "10.1210/jc.2018-00854",
language = "English",
volume = "103",
pages = "3792--3800",
journal = "Journal of Clinical Endocrinology and Metablism",
issn = "0021-972X",
publisher = "The Endocrine Society",
number = "10",

}

TY - JOUR

T1 - A cross-sectional and longitudinal analysis of trabecular bone score in adults with turner syndrome

AU - Nguyen, Hanh Huu

AU - Wong, Phillip

AU - Strauss, Boyd J

AU - Ebeling, Peter R

AU - Milat, Frances

AU - Vincent, Amanda

PY - 2018/10/1

Y1 - 2018/10/1

N2 - Context: Turner syndrome (TS) is associated with short stature, gonadal failure, and fractures. Spinal trabecular bone score (TBS) is a novel bone imaging modality that has not been evaluated in TS. Objective: To evaluate TBS in TS and its association with bone mineral density (BMD), prevalent fracture, and risk factors. Design and Setting: Longitudinal study of TS from a single tertiary hospital between 2006 and 2017. Patients or Other Participants: Fifty-eight subjects with TS aged 20 to 49 years who underwent dualenergy X-ray absorptiometry (DXA). Main Outcome Measures: TBS, DXA parameters, and prevalent fractures were investigated. Results: Normal, partially degraded, and degraded TBSs were observed in 39 (67%), 15 (26%), and four (7%) subjects, respectively. High rates of prescribed estrogen replacement therapy (ERT) with stable TBS and BMD were observed during follow-up. TBS was positively correlated with spine and femoral neck (FN) BMD and Z-scores (all P<0.05) and negatively correlated with age (20.004 per year; P = 0.014) and delay in ERT initiation inwomenwith primary amenorrhea (20.010 per year; P<0.001). Fractureswere present in 17 (31%) subjects. LowTBS had a significantly higher area under the receiver operator curve for predicting prevalent fracture than low bone mass at either the spine or FN (P < 0.05). Subjects with no history of fracture were more likely to have a normal TBS (P = 0.023). Conclusions: BMD and TBS can be preserved with early initiation and continued use of ERT. TBS may provide additional fracture risk prediction to standard DXA parameters in TS and needs to be validated in larger prospective studies.

AB - Context: Turner syndrome (TS) is associated with short stature, gonadal failure, and fractures. Spinal trabecular bone score (TBS) is a novel bone imaging modality that has not been evaluated in TS. Objective: To evaluate TBS in TS and its association with bone mineral density (BMD), prevalent fracture, and risk factors. Design and Setting: Longitudinal study of TS from a single tertiary hospital between 2006 and 2017. Patients or Other Participants: Fifty-eight subjects with TS aged 20 to 49 years who underwent dualenergy X-ray absorptiometry (DXA). Main Outcome Measures: TBS, DXA parameters, and prevalent fractures were investigated. Results: Normal, partially degraded, and degraded TBSs were observed in 39 (67%), 15 (26%), and four (7%) subjects, respectively. High rates of prescribed estrogen replacement therapy (ERT) with stable TBS and BMD were observed during follow-up. TBS was positively correlated with spine and femoral neck (FN) BMD and Z-scores (all P<0.05) and negatively correlated with age (20.004 per year; P = 0.014) and delay in ERT initiation inwomenwith primary amenorrhea (20.010 per year; P<0.001). Fractureswere present in 17 (31%) subjects. LowTBS had a significantly higher area under the receiver operator curve for predicting prevalent fracture than low bone mass at either the spine or FN (P < 0.05). Subjects with no history of fracture were more likely to have a normal TBS (P = 0.023). Conclusions: BMD and TBS can be preserved with early initiation and continued use of ERT. TBS may provide additional fracture risk prediction to standard DXA parameters in TS and needs to be validated in larger prospective studies.

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U2 - 10.1210/jc.2018-00854

DO - 10.1210/jc.2018-00854

M3 - Article

VL - 103

SP - 3792

EP - 3800

JO - Journal of Clinical Endocrinology and Metablism

JF - Journal of Clinical Endocrinology and Metablism

SN - 0021-972X

IS - 10

ER -