A 6-year follow-up of fracture incidence and volumetric bone mineral density development in girls with Turner syndrome
Abstract
Context: Patients with Turner syndrome (TS) are at risk for osteoporotic fractures. Objective: The aims of this study were to assess the incidence of clinically important fractures in girls with TS and prospectively describe the development of volumetric bone mineral density (BMD). Design: Peripheral quantitative computerized tomography (pQCT) of the radius every other year over the 6 years of observation. Setting: Government-funded university referral center. Participants: Thirty-two girls with TS, aged 6 to 16 years, were included in the analyses. Fracture incidence was compared with the data in the general population. Bone density and strength were compared with data from 185 healthy girls. Outcomes: The main clinical outcome was the fracture occurrence. The secondary outcomes were the changes in Z-scores of the bone parameters. Results: Three girls with TS sustained four fractures during 6 years of observation. The fracture rate in TS was not substantially higher than the downward-biased fracture-rate estimate from age-matched, healthy controls (P = 0.48). Whereas the trabecular BMD Z-score decreased with age (β estimate -0.21 ± 0.04, P < 0.001), total bone cross-sectional area correspondingly increased (+0.16 ± 0.04, P < 0.001), which led to normal bone strength. A positive history of incident fractures was not significantly associated with any of the pQCT-derived bone parameters. Conclusions: Current pediatric TS patients that are treated with growth hormone and estrogens are not at risk for osteoporotic fractures. Low BMD in TS may be counterweighted by enlarged bone radius, which leads to normal bone strength at the appendicular skeleton.
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Earlier initiation of HRT affects BMD with higher BMD/better bone quality/greater trabecular bone score.
“Earlier initiation of HRT does affect BMD with higher BMD/better bone quality/greater trabecular bone score”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [351]
The "dose effect" of the SHOX gene is associated with thinning of cortical bone and increased bone geometry at the distal radius.
“Although karyotype per se737 is not predictive of BMD, the “dose effect” of the SHOX gene is associated with thinning of cortical bone and increased bone geometry at the distal radius.351,738,739”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [351]
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References 41 · Cited by 30
- Authors
- Soucek, O., Schönau, E., Lebl, J., Willnecker, J., Hlavka, Z., & Sumnik, Z.
- Affiliations
- Department of Pediatrics, Second Medical Faculty, Charles University in Prague and Motol University Hospital, Prague, Czech Republic
- Department of Pediatrics, University Hospital Cologne and Cologne University, Cologne, Germany
- Charles University (CZ)
- Stratec (Germany) (DE)
- Department of Probability and Mathematical Statistics, Faculty of Mathematics and Physics, Charles University in Prague, Prague, Czech Republic
- Venue
- The Journal of Clinical Endocrinology & Metabolism
- Publisher
- The Endocrine Society
- Published
- 2017-12-28
- Volume
- 103
- Issue
- 3
- Pages
- 1188-1197
- ISSN
- 0021-972X (ISSN-L)
- 1945-7197
- OpenAlex
- W2778273483
- Cited by
- 30
- References
- 41
- Citations by year
- 2026: 1 · 2025: 3 · 2024: 2 · 2023: 2 · 2022: 3 · 2021: 4 · 2020: 7 · 2019: 4 · 2018: 4
- FWCI
- 4.51
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
- PubMed (publishedVersion)
- Kölner Universitäts PublikationsServer (Universität zu Köln) (publishedVersion)
- Topics
- Health Sciences › Medicine › Orthopedics and Sports Medicine › Bone health and osteoporosis research
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Clinical Aspects of Sex Determination and Chromosomal Abnormalities
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Sexual Differentiation and Disorders
- Keywords
- Turner syndrome, fracture incidence, volumetric bone mineral density, trabecular bone, bone cross-sectional area, bone strength, osteoporotic fractures, growth hormone therapy, estrogen therapy, pediatric girls, appendicular skeleton, peripheral quantitative computed tomography
- MeSH terms
- Adolescent, Child, Estrogens, Female, Follow-Up Studies, Humans, Radius, Risk Factors, Growth Hormone, Time Factors, Tomography, X-Ray Computed, Turner Syndrome, Bone Density, Incidence, Osteoporotic Fractures
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