Hydrocortisone misuse
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Cushing syndrome following administration error: case report A 6-year-old girl developed Cushing syndrome following misuse and administration error of hydrocortisone for salt-wasting classic congenital adrenal hyperplasia (SW-CAH) The girl, who had salt-wasting SW-CAH, presented at 6 years and 11 months with poor growth, increasing BMI, hirsutism, round facies and gastric ulcers. Molecular testing confirmed SW-CAH diagnosis. Her parents self made hydrocortisone suspension to deliver mean total daily dose of 4.3+/-1.8 mg/day. Concomitant medication included fludrocortisone. She had elevated BMI since age of 3 years, with a sharp BMI increase around the age of 6 years. Following presentation, all adrenal steroid concentrations including 17-hydroxyprogesterone, androstenedione and adrenocorticotropin were undetectable in the morning, 12 hours following her last hydrocortisone dose, which indicated chronic hypothalamic-pituitary-adrenal (HPA) axis suppression. Further anamnesis revealed that every morning her parents dissolved a 5mg hydrocortisone tablet in 5mL water and administered orally using a syringe. During the entire year prior to the presentation, her parents dissolved hydrocortisone 10mg in 10mL water, in order to administer her with the suspension, which coincided with rapid increase in BMI. Following presentation, a 6h cortisol pharmacokinetic study revealed low clearance at 58.8 dL/hr/m2. Cushing syndrome following misuse and administration error of hydrocortisone was suspected. The girl was advised to switch to a pharmacy compounded alcohol-free hydrocortisone 6.5 mg/m2/day suspension, divided in four doses. At the 1-year follow up visit, her Cushingoid features resolved. Of note, BMI decreased (68th percentile) and growth was at 10th percentile. The alcohol-free suspension allowed for more accurate hydrocortisone administration. HPA axis recovered, and clinical manifestations improved without notable change in the nominal hydrocortisone dose. Al-Rayess H, et al. Iatrogenic cushing syndrome in a girl with classic congenital adrenal hyperplasia treated with a homemade hydrocortisone solution. Hormone Research in 803505915 Paediatrics 93 (Suppl. 1): 99-100 (plus poster) abstr. 146, Jul 2020. Available from: URL: http://doi.org/10.1159/000509566 [abstract]
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Reactions 10 Oct 2020 No. 1825
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