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Continuous subcutaneous hydrocortisone infusion in pediatric primary adrenal insufficiency: a cohort study

Khadidja Fouatih1, Trouvin Marie-Agathe1, Lambert Anne-Sophie1,2

  • 1AP-HP, Université Paris-Saclay, Service Endocrinologie et diabète de l'enfant, Centre de Référence des Maladies Rares du Développement Génital (CRMR DEVGEN), Hôpital Bicêtre Paris-Saclay, Le Kremlin-Bicêtre, France.

Endocrine Connections
|January 6, 2026
PubMed

Insights

Continuous subcutaneous hydrocortisone infusion (CSHI) is a safe and effective treatment for pediatric primary adrenal insufficiency (PAI) when oral therapy fails. This method improves hormonal control and offers a more physiological cortisol delivery in children with PAI.

Area of Science:

  • Pediatric Endocrinology
  • Hormone Replacement Therapy
  • Adrenal Disorders

Background:

  • Primary adrenal insufficiency (PAI) in children, often due to congenital adrenal hyperplasia (CAH), presents treatment challenges.
  • Mimicking physiological cortisol rhythms is difficult with standard oral hydrocortisone due to its short half-life.
  • Continuous subcutaneous hydrocortisone infusion (CSHI) shows promise but is understudied in pediatric populations.

Purpose of the Study:

  • To assess the feasibility, safety, and clinical efficacy of CSHI in pediatric patients with PAI.
  • To evaluate CSHI as an alternative for children with PAI inadequately controlled by oral medications.

Main Methods:

  • A retrospective monocentric study involving 13 children and adolescents with PAI.
  • Patients were switched from oral hydrocortisone to CSHI due to poor disease control.
  • Hormonal and clinical parameters were monitored over a median follow-up of 48 months.

Main Results:

  • CSHI improved biochemical control, increasing morning cortisol and decreasing ACTH, 17-OHP, androstenedione, and testosterone.
  • Growth velocity and BMI remained stable; adrenal crises ceased in one patient.
  • Tumor volume in testicular adrenal rest tumors decreased or resolved in boys, and menstrual cycles resumed in an adolescent girl.
  • CSHI was well-tolerated with no major complications.

Conclusions:

  • CSHI is a viable and safe therapeutic option for pediatric PAI patients poorly managed with oral therapy.
  • The infusion provides more physiological cortisol delivery and enhances hormonal control.
  • Larger prospective studies are warranted to confirm efficacy and assess quality-of-life outcomes.
Abstract

Keywords:
congenital adrenal hyperplasiacontinuous subcutaneous hydrocortisone infusionprimary adrenal insufficiency

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