title: Results of the prolonged use of subcutaneous continuous infusion of hydrocortisone in a man with congenital adrenal hyperplasia.
abstract: This is a case report study of a young man with Congenital Adrenal Hyperplasia (CAH) who has been treated during 2 years by a subcutaneous continuous infusion hydrocortisone (SCIH) to optimize his treatment. Hydrocortisone was delivered via an insulin infusion device. We also studied the evolution of testicular adrenal rest tumors (TARTs) and the quality of life through SF36 survey. Four rates were determined, with a total of 47 mg per day. Biochemical parameters were normalized at 2 months. The SF36 questionnaire showed a progress of well-being. The weight decreased to 106 kg, that is, -5 kg (height: 1.71 m). Unfortunatly, there was no change of the TARTs. Two episodes of dermohypodermitis, with abscess at the infusion site, were observed. This case demonstrates the feasibility of prolonged SCIH therapy in patients with CAH, reporting positive effects on quality of life and on BMI.
authors.affiliation: Service d'Endocrinologie, Hôpital de la Cavale Blanche CHU de Brest, Boulevard Tanguy Prigent, 29609 Brest Cedex, France.
authors.forename: Emmanuel
authors.initials: E
authors.lastname: Sonnet
authors.forename: Nathalie
authors.initials: N
authors.lastname: Roudaut
authors.forename: Véronique
authors.initials: V
authors.lastname: Kerlan