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Cited by 11Growth Hormone and Insulin-like Growth FactorsGenetic and Clinical Aspects of Sex Determination and Chromosomal AbnormalitiesHypothalamic control of reproductive hormones

Normalization of puberty and adult height in girls with Turner syndrome: results of the Swedish growth hormone trials initiating transition into adulthood

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Kriström, Berit, Ankarberg-Lindgren, Carina, Barrenäs, Marie-Louise, Nilsson, Karl Olof, Albertsson-Wikland, Kerstin·2023·Frontiers in Endocrinology
DOI 10.3389/fendo.2023.1197897PMC PMC10389045
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Abstract

Objective To study the impact of GH dose and age at GH start in girls with Turner syndrome (TS), aiming for normal height and age at pubertal onset (PO) and at adult height (AH). However, age at diagnosis will limit treatment possibilities. Methods National multicenter investigator-initiated studies (TNR 87-052-01 and TNR 88-072) in girls with TS, age 3–16 years at GH start during year 1987–1998, with AH in 2003–2011. Of the 144 prepubertal girls with TS, 132 girls were followed to AH (intention to treat), while 43 girls reduced dose or stopped treatment prematurely, making n=89 for Per Protocol population. Age at GH start was 3–9 years (young; n=79) or 9–16 years (old; n=53). Treatment given were recombinant human (rh)GH (Genotropin® Kabi Peptide Hormones, Sweden) 33 or 67 µg/kg/day, oral ethinyl-estradiol (2/3) or transdermal 17β-estradiol (1/3), and, after age 11 years, mostly oxandrolone. Gain in heightSDS, AHSDS, and age at PO and at AH were evaluated. Results At GH start, heightSDS was −2.8 (versus non-TS girls) for all subgroups and mean age for young was 5.7 years and that of old was 11.6 years. There was a clear dose–response in both young and old TS girls; the mean difference was (95%CI) 0.66 (−0.91 to −0.26) and 0.57 (−1.0 to −0.13), respectively. The prepubertal gainSDS (1.3–2.1) was partly lost during puberty (−0.4 to −2.1). Age/heightSDS at PO ranged from 13 years/−0.42 for GH67young to 15.2 years/−1.47 for GH33old. At AH, GH67old group became tallest (17.2 years; 159.9 cm; −1.27 SDS; total gainSDS, 1.55) compared to GH67young group being least delayed (16.1 years; 157.1 cm; −1.73 SDS; total, 1.08). The shortest was the GH33young group (17.3 years; 153.7 cm: −2.28 SDS; total gainSDS, 0.53), and the most delayed was the GH33old group, (18.5 years; 156.5 cm; −1.82 SDS; total gainSDS, 0.98). Conclusion For both young and old TS girls, there was a GH-dose growth response, and for the young, there was less delayed age at PO and at AH. All four groups reached an AH within normal range, despite partly losing the prepubertal gain during puberty. Depending on age at diagnosis, low age at start with higher GH dose resulted in greater prepubertal height gain, permitting estrogen to start earlier at normal age and attaining normal AH at normal age, favoring physiological treatment and possibly also bone health, hearing, uterine growth and fertility, psychosocial wellbeing during adolescence, and the transition to adulthood.

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  1. [22]Nilsson K-O Albertsson-Wikland K Alm J Aronson S Gustafsson J Hagenäs L. Timing of oestrogen therapy in girls with turner syndrome: the Swedish experiences and a review. In: Saenger PP, editor. Otimizing health care for turner patients in the 21st century, proceedings. Naples, Italy: Elsevier (2000). p. 185–97.
  2. [55]Ivarsson S-A Carlsson A Bredberg A Alm J Aronsson S Gustafsson J. High prevalence of celiac disease in turner’s syndrome. Rev Ser Pediatrics (2001) 2(01): 12–4.
  3. [90]Wide Boman U Möller A Albertsson-Wikland K. Self-perception, behavior and social functioning in Swedish girls with turner syndrome: a population-based study. Göteborg psychol Rep Göteborg Univ Sweden (2000) 30(5): 1– 12.
  4. [120]Jansson P-O Barrenäs M-L Kriström B Hansson C Möller A Wilhelmsen-Landin K. The role of the gynecologist in the multidisciplinary management of girls and woman with turner syndrome. In: Balen A, editor. The multidisciplinary approach to the management of paediatric and adolescent gynaecology. Cambridge, UK: Cambridge University Press (2004). p. 242–52.
  5. [121]Albertsson Wikland K Ranke M, editors. Research and clinical aspects. The 4th international symposium on turner syndrome turner syndrome in a life span perspective. Gothenburg: Excerpta Medica ICS (1995).

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Authors
Kriström, B., Ankarberg-Lindgren, C., Barrenäs, M.-L., Nilsson, K. O., & Albertsson-Wikland, K.
Affiliations
  • Umeå University (SE)
  • University of Gothenburg (SE)
  • Lund University (SE)
Corresponding author
Albertsson-Wikland, Kerstin
ORCID
Venue
Frontiers in Endocrinology
Publisher
Frontiers Media SA
Published
2023-07-17
Volume
14
Pages
1197897-1197897
ISSN
1664-2392 (ISSN-L)
DOI
10.3389/fendo.2023.1197897
PMC
PMC10389045
OpenAlex
W4384488655
Cited by
11
References
117
Citations by year
2026: 1 · 2025: 2 · 2024: 7 · 2023: 1
FWCI
2.26
Type
journal article
Language
en
Open access
Licence
Locations
  • Frontiers in Endocrinology (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • KTH Publication Database DiVA (KTH Royal Institute of Technology) (submittedVersion, other-oa)
  • DiVA at Umeå University (Umeå University) (submittedVersion, other-oa)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
  • Lund University Publications (Lund University) (submittedVersion)
Funders
Vetenskapsrådet
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
  • Health Sciences › Medicine › Reproductive Medicine › Hypothalamic control of reproductive hormones
Keywords
Turner syndrome, growth hormone therapy, adult height, puberty onset, recombinant human growth hormone, dose-response relationship, age at treatment initiation, prepubertal girls, puberty suppression, estradiol, oxandrolone, height SDS, coming of age
MeSH terms
Adolescent, Adult, Body Height, Child, Child, Preschool, Estradiol, Female, Humans, Puberty, Growth Hormone, Sweden, Turner Syndrome, Human Growth Hormone

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