Authors
Gülay Karagüzel, Esra Baki Erkul, Yüksel Aliyazıcıoğlu, Bircan Sönmez, Nazım Ercüment Beyhun
Published in
European journal of pediatrics. Volume 185. Issue 8. Jul 20, 2026. Epub Jul 20, 2026.
Abstract
Serum sclerostin levels as a negative regulator of bone formation in children with congenital adrenal hyperplasia (CAH) have not been evaluated to date. The study aimed to investigate serum sclerostin and osteoprotegerin levels in children with CAH. Thirty-one patients with CAH (mean age 11.6 ± 3.7 years) taking glucocorticoids for at least 5 years, and age- and gender-matched controls were included in this cross-sectional study. Serum levels of sclerostin, osteoprotegerin, and standard bone markers were assessed. Bone mineral density (BMD) was measured using dual-energy X-ray absorptiometry. The factors influencing serum sclerostin and osteoprotegerin levels were identified using generalized linear modeling. In healthy controls, being pubertal was associated with lower serum sclerostin levels, whereas pubertal patients did not demonstrate the expected decline in sclerostin levels. Osteoprotegerin levels were higher in patients than in controls (p < 0.001). Serum 17-hydroxy progesterone exerted a significant negative effect on osteoprotegerin and a positive effect on sclerostin. Cumulative glucocorticoid dose, vitamin D, pubertal status, and sex had no significant effect on sclerostin or osteoprotegerin levels in patients. The BMD Z-scores exceeded - 1 in 80% of both patient and controls.
The present study is the first to investigate the role of sclerostin in bone metabolism in patients with CAH, revealing sclerostin- and osteoprotegerin-mediated alterations in bone metabolism in this population. These alterations, along with their associations with serum 17-hydroxy progesterone levels, may be related to disease control status. Furthermore, the absence of the expected pubertal decline in sclerostin levels may adversely affect peak bone mass acquisition during growth.
• Promoting bone mass accumulation during puberty is important for lifelong skeletal health. The long-term effect of glucocorticoid treatment on bone health in CAH patients is still unclear.
• The present study provides evidence for sclerostin- and osteoprotegerin-mediated alterations in bone metabolism in children with CAH, potentially influenced by 17-OHP, and these findings may inform future research in this field.
PMID:
42474852
Bibliographic data and abstract were imported from PubMed on 20 Jul 2026.
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