Growth and adult height in girls with Turner syndrome following IGF-1 titrated growth hormone treatment
Abstract
CONTEXT: Girls with Turner syndrome (TS) suffer linear growth failure, and TS is a registered indication for growth hormone (GH) treatment. GH is classically dosed according to body weight, and serum insulin-like growth factor-1 (IGF-1) concentrations are recommended to be kept within references according to international guidelines. OBJECTIVE: To assess the effect of long-term GH treatment in girls with TS following GH dosing by IGF-1 titration. DESIGN AND SETTING: A retrospective, real-world evidence, observational study consisting of data collected in a single tertiary center from 1991 to 2018. PATIENTS: A cohort of 63 girls with TS treated with GH by IGF-1 titration with a median duration of 6.7 years (interquartile range [IQR]: 3.4-9.7 years). MAIN OUTCOME MEASURES: Longitudinal measurements of height, IGF-1, and adult height (AH) following GH treatment were evaluated and compared between the different karyotypes (45,X, 45,X/46,XX, or miscellaneous). RESULTS: Using GH dose titration according to IGF-1, only 6% of girls with TS had supranormal IGF-1 levels. Median dose was 33 µg/kg/day (IQR: 28-39 µg/kg/day) with no difference between the karyotype groups. AH was reached for 73% who attained a median AH of 1.25 standard deviation score (SDS) for age specific TS references (IQR: 0.64-1.50 SDS), and a median gain in height (ΔHSDS: AH SDS minus baseline height SDS of TS references) of 0.50 SDS, equal to 3.2 cm (SD 7.68) for all karyotypes. CONCLUSION: Our real-world evidence study suggested that titration of GH dose to keep IGF-1 levels within the normal range resulted in a lower AH gain than in studies where a fixed dose was used.
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Observational data show reduced efficacy when prescribed GH doses are lower than recommended doses.
“Observational data185-188 show reduced efficacy when prescribed doses are lower than recommended doses.”
From Clinical practice guidelines for the care of girls and women with Turner syndrome. (2024) · cites this paper as [187]
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- Authors
- Cleemann Wang, A., Hagen, C. P., Nedaeifard, L., Juul, A., & Jensen, R. B.
- Affiliations
- Department of Growth and Reproduction, Rigshospitalet, University of Copenhagen, Copenhagen, Denmark
- University of Copenhagen (DK)
- Venue
- The Journal of Clinical Endocrinology & Metabolism
- Publisher
- The Endocrine Society
- Published
- 2020-05-18
- Volume
- 105
- Issue
- 8
- Pages
- 2566-2574
- ISSN
- 0021-972X (ISSN-L)
- 1945-7197
- OpenAlex
- W3027384300
- Cited by
- 12
- References
- 29
- Citations by year
- 2026: 2 · 2025: 2 · 2024: 1 · 2023: 5 · 2022: 2
- FWCI
- 1.32
- Type
- journal article
- Language
- en
- Open access
- closed
- Licence
- academic.oup.com/journals/pages/open_access/funder_policies/chorus/standard_publication_model (vor)
- Locations
- The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
- PubMed (publishedVersion)
- Research at the University of Copenhagen (University of Copenhagen) (submittedVersion)
- Funders
- Rigshospitalet Research Foundation
- Topics
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Growth Hormone and Insulin-like Growth Factors
- 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, growth hormone therapy, adult height, karyotype, 45,X/46,XX mosaicism, height gain, dose adjustment
- MeSH terms
- Adolescent, Body Height, Child, Dose-Response Relationship, Drug, Female, Humans, Insulin-Like Growth Factor I, Karyotyping, Longitudinal Studies, Reference Values, Retrospective Studies, Turner Syndrome, Treatment Outcome, Human Growth Hormone, Drug Dosage Calculations, Karyotype
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