{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,31]],"date-time":"2026-10-31T05:36:32Z","timestamp":1793424992175,"version":"4.5.0"},"reference-count":54,"publisher":"The Endocrine Society","issue":"2","license":[{"start":{"date-parts":[[2019,11,21]],"date-time":"2019-11-21T00:00:00Z","timestamp":1574294400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,2,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>To establish short- and long-term adverse outcome frequencies related to a late diagnosis of congenital adrenal hyperplasia (CAH) in the absence of newborn screening (NBS) and to determine respective treatment costs, which have never been reported.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Design<\/jats:title>\n                  <jats:p>A retrospective analysis of a CAH cohort diagnosed without NBS.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>We evaluated medical record data concerning 195 patients (141 females) diagnosed with CAH through clinical suspicion and confirmed using hormonal and CYP21A2 analysis, who were followed from 1980 to 2016 at Sao Paulo University. We measured mortality, dehydration, mental impairment frequencies, and hospitalization length outcomes in the salt-wasting form; the frequency of genetic females raised as males in both forms, frequency of depot GnRh analog (GnRha) and GH therapies in the simple virilizing form, and related outcome costs were calculated.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Mortality rates and associated costs, varying from 10% to 26% and from $2,239,744.76 to $10,271,591.25, respectively, were calculated using the Brazilian yearly live-births rate, estimated productive life years, and gross domestic product. In the salt-wasting form, 76% of patients were hospitalized, 8.6% were mentally impaired, and 3% of females were raised as males (total cost, $86,230\/salt-wasting patient). GnRha and growth hormone were used for 28% and 14% of simple virilizing patients, respectively, and 18% of females were raised as males (preventable cost, $4232.74\/simple virilizing patient).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>A late CAH diagnosis leads to high mortality and morbidity rates, notably increasing public health costs, and may result in physical and psychological damage that is not easily measurable.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/jendso\/bvz013","type":"journal-article","created":{"date-parts":[[2019,11,21]],"date-time":"2019-11-21T12:00:21Z","timestamp":1574337621000},"source":"Crossref","is-referenced-by-count":15,"title":["Adverse Outcomes and Economic Burden of Congenital Adrenal Hyperplasia Late Diagnosis in the Newborn Screening Absence"],"prefix":"10.1210","volume":"4","author":[{"given":"Mirela Costa De","family":"Miranda","sequence":"first","affiliation":[{"name":"Developmental Endocrinology Unit, Laborat\u00f3rio de Horm\u00f4nios e Gen\u00e9tica Molecular\/LIM42, Hospital das Cl\u00ednicas da Faculdade de Medicina da Universidade de S\u00e3o Paulo, S\u00e3o Paulo, Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Luciana Bertocco de Paiva","family":"Haddad","sequence":"additional","affiliation":[{"name":"Liver and Gastrointestinal Transplant Division, Department of Gastroenterology, Hospital das Cl\u00ednicas da Faculdade de Medicina da Universidade de S\u00e3o Paulo, S\u00e3o Paulo, Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guiomar","family":"Madureira","sequence":"additional","affiliation":[{"name":"Developmental Endocrinology Unit, Laborat\u00f3rio de Horm\u00f4nios e Gen\u00e9tica Molecular\/LIM42, Hospital das Cl\u00ednicas da Faculdade de Medicina da Universidade de S\u00e3o Paulo, S\u00e3o Paulo, Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Berenice Bilharinho de","family":"Mendonca","sequence":"additional","affiliation":[{"name":"Developmental Endocrinology Unit, Laborat\u00f3rio de Horm\u00f4nios e Gen\u00e9tica Molecular\/LIM42, Hospital das Cl\u00ednicas da Faculdade de Medicina da Universidade de S\u00e3o Paulo, S\u00e3o Paulo, Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tania A S S","family":"Bachega","sequence":"additional","affiliation":[{"name":"Developmental Endocrinology Unit, Laborat\u00f3rio de Horm\u00f4nios e Gen\u00e9tica Molecular\/LIM42, Hospital das Cl\u00ednicas da Faculdade de Medicina da Universidade de S\u00e3o Paulo, S\u00e3o Paulo, Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2019,11,21]]},"reference":[{"issue":"5","key":"2021121116404545800_CIT0001","doi-asserted-by":"crossref","first-page":"400","DOI":"10.1055\/s-0032-1324724","article-title":"Congenital adrenal hyperplasia due to 21 hydroxylase deficiency: from birth to adulthood","volume":"30","author":"White","year":"2012","journal-title":"Semin Reprod Med."},{"issue":"9","key":"2021121116404545800_CIT0002","doi-asserted-by":"crossref","first-page":"490","DOI":"10.1038\/nrendo.2009.148","article-title":"Neonatal screening for congenital adrenal hyperplasia","volume":"5","author":"White","year":"2009","journal-title":"Nat Rev Endocrinol."},{"issue":"4","key":"2021121116404545800_CIT0003","doi-asserted-by":"crossref","first-page":"480","DOI":"10.1111\/cen.13292","article-title":"Neonatal 17-hydroxyprogesterone levels adjusted according to age at sample collection and birthweight improve the efficacy of congenital adrenal hyperplasia newborn screening","volume":"86","author":"Hayashi","year":"2017","journal-title":"Clin Endocrinol (Oxf)."},{"issue":"5","key":"2021121116404545800_CIT0004","doi-asserted-by":"crossref","first-page":"455","DOI":"10.1515\/JPEM.2008.21.5.455","article-title":"The actual incidence of congenital adrenal hyperplasia in Brazil may not be as high as inferred\u2013an estimate based on a public neonatal screening program in the state of Goi\u00e1s","volume":"21","author":"Silveira","year":"2008","journal-title":"J Pediatr Endocrinol Metab."},{"issue":"7","key":"2021121116404545800_CIT0005","doi-asserted-by":"crossref","first-page":"765","DOI":"10.1590\/0004-2730000003310","article-title":"Ten-year evaluation of a neonatal screening program for congenital adrenal hyperplasia","volume":"58","author":"Nascimento","year":"2014","journal-title":"Arq Bras Endocrinol 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