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International practice of corticosteroid replacement therapy in congenital adrenal hyperplasia: data from the I-CAH

Irina Bacila1, Nicole Freeman1, Eleni Daniel1

  • 1Department of Oncology and Metabolism, University of Sheffield, Sheffield, UK.

European Journal of Endocrinology
|January 18, 2021
PubMed

Insights

Hormone replacement therapy for congenital adrenal hyperplasia (CAH) varies significantly worldwide. This study found international differences in glucocorticoid and mineralocorticoid use, with a trend toward higher doses in children with CAH.

Area of Science:

  • Pediatric Endocrinology
  • Genetics and Genetic Diseases
  • Pharmacology and Therapeutics

Background:

  • Congenital adrenal hyperplasia (CAH) management lacks a unified global approach despite existing guidelines.
  • Hormone replacement therapy is crucial for managing CAH, impacting patient outcomes.

Purpose of the Study:

  • To investigate geographical and temporal variations in glucocorticoid and mineralocorticoid replacement therapy for CAH.
  • To analyze treatment patterns in children and adolescents with classic 21-hydroxylase deficiency.

Main Methods:

  • Retrospective analysis of data from the International-CAH Registry, including 31 centers across 16 countries.
  • Inclusion of 461 patients (0-18 years) with classic 21-hydroxylase deficiency, followed between 1982 and 2018.
  • Analysis of 4174 patient visits, examining type, dose, and timing of steroid hormone replacement.

Main Results:

  • Hydrocortisone was the most common glucocorticoid (87.6%).
  • Glucocorticoid doses varied by age group, with higher doses in older children (12-18 years).
  • Doses decreased after 2010 for younger children (0-8 years), and fludrocortisone use decreased with age, showing significant international variations in all aspects of therapy.

Conclusions:

  • International variations in hormone replacement therapy for CAH are evident.
  • A trend towards higher steroid doses in pediatric CAH patients was observed.
  • The findings highlight the need for standardized treatment protocols in CAH management.
Abstract

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