{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,7]],"date-time":"2026-10-07T01:52:27Z","timestamp":1791337947552,"version":"4.3.1"},"reference-count":20,"publisher":"The Endocrine Society","issue":"3","license":[{"start":{"date-parts":[[2020,9,5]],"date-time":"2020-09-05T00:00:00Z","timestamp":1599264000000},"content-version":"vor","delay-in-days":1,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"European Commission Framework 7 Programme","award":["281654"],"award-info":[{"award-number":["281654"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,3,8]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>Children with congenital adrenal hyperplasia (CAH) and adrenal insufficiency (AI) require daily hydrocortisone replacement with accurate dosing.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>Prospective study of efficacy and safety of hydrocortisone granules in children with AI and CAH monitored by 17-OHP (17-hydroxyprogesterone) saliva profiles.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>Seventeen children with CAH (9 male) and 1 with hypopituitarism (male), aged from birth to 6 years, had their hydrocortisone medication changed from pharmacy compounded capsules to hydrocortisone granules. Patients were followed prospectively for 2 years. In children with CAH, the therapy was adjusted by 17-OHP salivary profiles every 3 months. The following parameters were recorded: hydrocortisone dose, height, weight, pubertal status, adverse events, and incidence of adrenal crisis.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>The study medication was given thrice daily, and the median duration of treatment (range) was 795 (1\u2013872) days, with 150 follow-up visits. Hydrocortisone doses were changed on 40\/150 visits, with 32 based on salivary measurements and 8 on serum 17-OHP levels. The median daily mg\/m2 hydrocortisone dose (range) at study entry for the different age groups 2\u20138 years, 1 month to 2 years, &amp;lt;28 days was 11.9 (7.2\u201315.5), 9.9 (8.6\u201312.2), and 12.0 (11.1\u201329.5), respectively, and at end of the study was 10.2 (7.0\u201314.4), 9.8 (8.9\u201313.1), and 8.6 (8.2\u201313.7), respectively. There were no trends for accelerated or reduced growth. No adrenal crises were observed despite 193 treatment-emergent adverse events, which were mainly common childhood illnesses.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Interpretation<\/jats:title>\n                  <jats:p>This first prospective study of glucocorticoid treatment in children with AI and CAH demonstrates that accurate dosing and monitoring from birth results in hydrocortisone doses at the lower end of the recommended dose range and normal growth, without occurrence of adrenal crises.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgaa626","type":"journal-article","created":{"date-parts":[[2020,9,5]],"date-time":"2020-09-05T00:52:29Z","timestamp":1599267149000},"page":"e1433-e1440","source":"Crossref","is-referenced-by-count":36,"title":["A Prospective Study of Children Aged 0\u20138 Years with CAH and Adrenal Insufficiency Treated with Hydrocortisone Granules"],"prefix":"10.1210","volume":"106","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3892-0577","authenticated-orcid":false,"given":"Uta","family":"Neumann","sequence":"first","affiliation":[{"name":"Charit\u00e9 Universitaetsmedizin Berlin, Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katarina","family":"Braune","sequence":"additional","affiliation":[{"name":"Charit\u00e9 Universitaetsmedizin Berlin, Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin J","family":"Whitaker","sequence":"additional","affiliation":[{"name":"Diurnal Ltd., Cardiff, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Susanna","family":"Wiegand","sequence":"additional","affiliation":[{"name":"Charit\u00e9 Universitaetsmedizin Berlin, Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heiko","family":"Krude","sequence":"additional","affiliation":[{"name":"Charit\u00e9 Universitaetsmedizin Berlin, Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John","family":"Porter","sequence":"additional","affiliation":[{"name":"Diurnal Ltd., Cardiff, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dena","family":"Digweed","sequence":"additional","affiliation":[{"name":"Diurnal Ltd., Cardiff, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernard","family":"Voet","sequence":"additional","affiliation":[{"name":"Diurnal Ltd., Cardiff, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard J M","family":"Ross","sequence":"additional","affiliation":[{"name":"University of Sheffield, Sheffield, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Oliver","family":"Blankenstein","sequence":"additional","affiliation":[{"name":"Charit\u00e9 Universitaetsmedizin Berlin, Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2020,9,4]]},"reference":[{"issue":"9935","key":"2021122117010527600_CIT0001","doi-asserted-by":"crossref","first-page":"2152","DOI":"10.1016\/S0140-6736(13)61684-0","article-title":"Adrenal insufficiency","volume":"383","author":"Charmandari","year":"2014","journal-title":"Lancet."},{"issue":"9","key":"2021122117010527600_CIT0002","doi-asserted-by":"crossref","first-page":"4133","DOI":"10.1210\/jc.2009-2631","article-title":"Congenital adrenal hyperplasia due to steroid 21-hydroxylase deficiency: an Endocrine Society clinical practice guideline","volume":"95","author":"Speiser","year":"2010","journal-title":"J Clin Endocrinol Metab."},{"issue":"1","key":"2021122117010527600_CIT0003","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1210\/jc.2017-01595","article-title":"Iatrogenic Cushing syndrome in a child with congenital adrenal hyperplasia: erroneous compounding of hydrocortisone","volume":"103","author":"Barillas","year":"2018","journal-title":"J Clin Endocrinol Metab."},{"issue":"4","key":"2021122117010527600_CIT0004","doi-asserted-by":"crossref","first-page":"1681","DOI":"10.1210\/jc.2014-4060","article-title":"Development and testing in healthy adults of oral hydrocortisone granules with taste masking for the treatment of neonates and infants with adrenal insufficiency","volume":"100","author":"Whitaker","year":"2015","journal-title":"J Clin Endocrinol Metab."},{"issue":"1","key":"2021122117010527600_CIT0005","doi-asserted-by":"crossref","first-page":"66","DOI":"10.1111\/cen.13876","article-title":"Accuracy of hydrocortisone dose administration via nasogastric tube","volume":"90","author":"Daniel","year":"2019","journal-title":"Clin Endocrinol (Oxf)."},{"issue":"1-2","key":"2021122117010527600_CIT0006","doi-asserted-by":"crossref","first-page":"57","DOI":"10.1016\/j.ijpharm.2018.04.054","article-title":"How close is the dose? Manipulation of 10\u202fmg hydrocortisone tablets to provide appropriate doses to children","volume":"545","author":"Watson","year":"2018","journal-title":"Int J Pharm."},{"issue":"2","key":"2021122117010527600_CIT0007","doi-asserted-by":"crossref","first-page":"239","DOI":"10.1530\/EJE-17-0248","article-title":"Quality of compounded hydrocortisone capsules used in the treatment of children","volume":"177","author":"Neumann","year":"2017","journal-title":"Eur J Endocrinol."},{"key":"2021122117010527600_CIT0008","first-page":"31","article-title":"Hydrocortisone granules with taste masking are well absorbed and tolerated in neonates, infants & children with adrenal insufficiency","volume":"88","author":"Neumann","year":"2017","journal-title":"Horm Res Paediat."},{"issue":"3","key":"2021122117010527600_CIT0009","doi-asserted-by":"crossref","first-page":"119","DOI":"10.1080\/17446651.2018.1455496","article-title":"Immediate-release granule formulation of hydrocortisone, Alkindi\u00ae, for treatment of paediatric adrenal insufficiency (Infacort Development Programme)","volume":"13","author":"Porter","year":"2018","journal-title":"Expert Rev Endocrinol Metab."},{"issue":"1","key":"2021122117010527600_CIT0010","doi-asserted-by":"crossref","first-page":"21","DOI":"10.1111\/cen.13447","article-title":"Absorption and tolerability of taste-masked hydrocortisone granules in neonates, infants and children under 6 years of age with adrenal insufficiency","volume":"88","author":"Neumann","year":"2018","journal-title":"Clin Endocrinol (Oxf)."},{"key":"2021122117010527600_CIT0011","author":"Neuhauser"},{"issue":"4","key":"2021122117010527600_CIT0012","first-page":"180","article-title":"The calculation of target height reconsidered","volume":"59","author":"Hermanussen","year":"2003","journal-title":"Horm Res."},{"issue":"3","key":"2021122117010527600_CIT0013","doi-asserted-by":"crossref","first-page":"R115","DOI":"10.1530\/EJE-14-0824","article-title":"Extensive expertise in endocrinology. 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