Neonatal screening for congenital adrenal hyperplasia in Southern Brazil: a population based study with 108,409 infants
Congenital adrenal hyperplasia (CAH) is an autosomal recessive disorder associated with inborn errors of steroid metabolism. 21-hydroxylase enzyme deficiency occurs in 90 to 95% of all cases of CAH, with accumulation of 17 hydroxyprogesterone (17-OHP). Early diagnosis of CAH based on newborn screeni...
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description | Congenital adrenal hyperplasia (CAH) is an autosomal recessive disorder associated with inborn errors of steroid metabolism. 21-hydroxylase enzyme deficiency occurs in 90 to 95% of all cases of CAH, with accumulation of 17 hydroxyprogesterone (17-OHP). Early diagnosis of CAH based on newborn screening is possible before the development of symptoms and allows proper treatment, correct sex assignment, and reduced mortality rates. This study describes the results obtained in the first year of a public CAH screening program in the state of Rio Grande do Sul, Brazil.
We reviewed the screening database in search of babies with suspected CAH, that is, altered birth-weight adjusted 17-OHP values at screening. The following data were analyzed for this population: screening 17-OHP values, retest 17-OHP values, serum 17-OHP values for those with confirmed CAH on retest, maternal and newborn data, and family history of CAH. For the screening program, 17-OHP levels are determined on dried blood spots obtained in filter paper with GSP solid phase time-resolved immunofluorescence.
Of 108,409 newborns screened, eight were diagnosed with CAH (four males, four females). The incidence of CAH in the state was 1:13,551. Six cases were identified as classic salt-wasting CAH and two were cases of virilizing CAH. The positive predictive value (PPV) of the initial screening (before diagnostic confirmation) was 1.6%. The overall rate of false positive results was 0.47%. The number of false positive results was higher among newborns with birth weight |
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We reviewed the screening database in search of babies with suspected CAH, that is, altered birth-weight adjusted 17-OHP values at screening. The following data were analyzed for this population: screening 17-OHP values, retest 17-OHP values, serum 17-OHP values for those with confirmed CAH on retest, maternal and newborn data, and family history of CAH. For the screening program, 17-OHP levels are determined on dried blood spots obtained in filter paper with GSP solid phase time-resolved immunofluorescence.
Of 108,409 newborns screened, eight were diagnosed with CAH (four males, four females). The incidence of CAH in the state was 1:13,551. Six cases were identified as classic salt-wasting CAH and two were cases of virilizing CAH. The positive predictive value (PPV) of the initial screening (before diagnostic confirmation) was 1.6%. The overall rate of false positive results was 0.47%. The number of false positive results was higher among newborns with birth weight < 2000 g.
The present results support the need for CAH screening by the public health care system in the state, and show that the strategy adopted is adequate. PPV and false positive results were similar to those reported for other states of Brazil with similar ethnic backgrounds.</description><identifier>ISSN: 1471-2431</identifier><identifier>EISSN: 1471-2431</identifier><identifier>DOI: 10.1186/s12887-016-0772-x</identifier><identifier>PMID: 28095810</identifier><language>eng</language><publisher>England: BioMed Central Ltd</publisher><subject>17-alpha-Hydroxyprogesterone - blood ; Adrenal Hyperplasia, Congenital - blood ; Adrenal Hyperplasia, Congenital - diagnosis ; Adrenal Hyperplasia, Congenital - epidemiology ; Adrenogenital syndrome ; Analysis ; Babies ; Biomarkers - blood ; Birth weight ; Brazil - epidemiology ; Diagnosis ; Early Diagnosis ; False Positive Reactions ; Female ; Health aspects ; Humans ; Hyperplasia ; Incidence ; Infant, Newborn ; Infants (Newborn) ; Male ; Medical screening ; Neonatal Screening - methods ; Newborn babies ; Pilot projects ; Population-based studies ; Predictive Value of Tests ; Retrospective Studies ; Steroids</subject><ispartof>BMC pediatrics, 2017-01, Vol.17 (1), p.22-22, Article 22</ispartof><rights>COPYRIGHT 2017 BioMed Central Ltd.</rights><rights>Copyright BioMed Central 2017</rights><rights>The Author(s). 2017</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c525t-22e6d4adf37bbd5eb6fa87953c4d7f0df60230f48d43e15dfe4d40b4e9db96a13</citedby><cites>FETCH-LOGICAL-c525t-22e6d4adf37bbd5eb6fa87953c4d7f0df60230f48d43e15dfe4d40b4e9db96a13</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5240440/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5240440/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,724,777,781,861,882,27905,27906,53772,53774</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/28095810$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Kopacek, Cristiane</creatorcontrib><creatorcontrib>de Castro, Simone Martins</creatorcontrib><creatorcontrib>Prado, Mayara Jorgens</creatorcontrib><creatorcontrib>da Silva, Claudia Maria Dornelles</creatorcontrib><creatorcontrib>Beltrão, Luciana Amorim</creatorcontrib><creatorcontrib>Spritzer, Poli Mara</creatorcontrib><title>Neonatal screening for congenital adrenal hyperplasia in Southern Brazil: a population based study with 108,409 infants</title><title>BMC pediatrics</title><addtitle>BMC Pediatr</addtitle><description>Congenital adrenal hyperplasia (CAH) is an autosomal recessive disorder associated with inborn errors of steroid metabolism. 21-hydroxylase enzyme deficiency occurs in 90 to 95% of all cases of CAH, with accumulation of 17 hydroxyprogesterone (17-OHP). Early diagnosis of CAH based on newborn screening is possible before the development of symptoms and allows proper treatment, correct sex assignment, and reduced mortality rates. This study describes the results obtained in the first year of a public CAH screening program in the state of Rio Grande do Sul, Brazil.
We reviewed the screening database in search of babies with suspected CAH, that is, altered birth-weight adjusted 17-OHP values at screening. The following data were analyzed for this population: screening 17-OHP values, retest 17-OHP values, serum 17-OHP values for those with confirmed CAH on retest, maternal and newborn data, and family history of CAH. For the screening program, 17-OHP levels are determined on dried blood spots obtained in filter paper with GSP solid phase time-resolved immunofluorescence.
Of 108,409 newborns screened, eight were diagnosed with CAH (four males, four females). The incidence of CAH in the state was 1:13,551. Six cases were identified as classic salt-wasting CAH and two were cases of virilizing CAH. The positive predictive value (PPV) of the initial screening (before diagnostic confirmation) was 1.6%. The overall rate of false positive results was 0.47%. The number of false positive results was higher among newborns with birth weight < 2000 g.
The present results support the need for CAH screening by the public health care system in the state, and show that the strategy adopted is adequate. PPV and false positive results were similar to those reported for other states of Brazil with similar ethnic backgrounds.</description><subject>17-alpha-Hydroxyprogesterone - blood</subject><subject>Adrenal Hyperplasia, Congenital - blood</subject><subject>Adrenal Hyperplasia, Congenital - diagnosis</subject><subject>Adrenal Hyperplasia, Congenital - epidemiology</subject><subject>Adrenogenital syndrome</subject><subject>Analysis</subject><subject>Babies</subject><subject>Biomarkers - blood</subject><subject>Birth weight</subject><subject>Brazil - epidemiology</subject><subject>Diagnosis</subject><subject>Early Diagnosis</subject><subject>False Positive Reactions</subject><subject>Female</subject><subject>Health aspects</subject><subject>Humans</subject><subject>Hyperplasia</subject><subject>Incidence</subject><subject>Infant, Newborn</subject><subject>Infants (Newborn)</subject><subject>Male</subject><subject>Medical screening</subject><subject>Neonatal Screening - methods</subject><subject>Newborn babies</subject><subject>Pilot projects</subject><subject>Population-based studies</subject><subject>Predictive Value of Tests</subject><subject>Retrospective Studies</subject><subject>Steroids</subject><issn>1471-2431</issn><issn>1471-2431</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><recordid>eNptUktv1DAYjBCIlsIP4IIsISEOpNiO7TgckErFS6rgAJwtJ_68ceW1g53QLr8eL1vKLkI-fH7MjKWZqarHBJ8SIsXLTKiUbY2JqHHb0vr6TnVMWEtqyhpyd29_VD3I-RJj0kom7ldHVOKOS4KPq6tPEIOetUd5SADBhRWyMaEhhlU5bR-0SRDKHDcTpMnr7DRyAX2JyzxCCuhN0j-df4U0muK0eD27GFCvMxiU58Vs0JWbR0SwfMFwV5hWhzk_rO5Z7TM8upkn1bd3b7-ef6gvPr__eH52UQ-c8rmmFIRh2tim7XvDoRdWy7bjzcBMa7GxAtMGWyYNa4BwY4EZhnsGnek7oUlzUr3e6U5LvwYzQJiT9mpKbq3TRkXt1OFLcKNaxR-KU4YZw0Xg-Y1Ait8XyLNauzyA9zpAXLIqORAuJCOyQJ_-A72MSyrW_UYxQTmR3V_USntQxY5Y_h22ouqMtSUUITgrqNP_oMoysHYlHLCu3B8Qnu0RRtB-HnP0yzaNfAgkO-CQYs4J7K0ZBKttrdSuVqrUSm1rpa4L58m-i7eMPz1qfgER9ch5</recordid><startdate>20170117</startdate><enddate>20170117</enddate><creator>Kopacek, Cristiane</creator><creator>de Castro, Simone Martins</creator><creator>Prado, Mayara Jorgens</creator><creator>da Silva, Claudia Maria Dornelles</creator><creator>Beltrão, Luciana Amorim</creator><creator>Spritzer, Poli Mara</creator><general>BioMed Central Ltd</general><general>BioMed Central</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20170117</creationdate><title>Neonatal screening for congenital adrenal hyperplasia in Southern Brazil: a population based study with 108,409 infants</title><author>Kopacek, Cristiane ; de Castro, Simone Martins ; Prado, Mayara Jorgens ; da Silva, Claudia Maria Dornelles ; Beltrão, Luciana Amorim ; Spritzer, Poli Mara</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c525t-22e6d4adf37bbd5eb6fa87953c4d7f0df60230f48d43e15dfe4d40b4e9db96a13</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>17-alpha-Hydroxyprogesterone - blood</topic><topic>Adrenal Hyperplasia, Congenital - blood</topic><topic>Adrenal Hyperplasia, Congenital - diagnosis</topic><topic>Adrenal Hyperplasia, Congenital - epidemiology</topic><topic>Adrenogenital syndrome</topic><topic>Analysis</topic><topic>Babies</topic><topic>Biomarkers - blood</topic><topic>Birth weight</topic><topic>Brazil - epidemiology</topic><topic>Diagnosis</topic><topic>Early Diagnosis</topic><topic>False Positive Reactions</topic><topic>Female</topic><topic>Health aspects</topic><topic>Humans</topic><topic>Hyperplasia</topic><topic>Incidence</topic><topic>Infant, Newborn</topic><topic>Infants (Newborn)</topic><topic>Male</topic><topic>Medical screening</topic><topic>Neonatal Screening - methods</topic><topic>Newborn babies</topic><topic>Pilot projects</topic><topic>Population-based studies</topic><topic>Predictive Value of Tests</topic><topic>Retrospective Studies</topic><topic>Steroids</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Kopacek, Cristiane</creatorcontrib><creatorcontrib>de Castro, Simone Martins</creatorcontrib><creatorcontrib>Prado, Mayara Jorgens</creatorcontrib><creatorcontrib>da Silva, Claudia Maria Dornelles</creatorcontrib><creatorcontrib>Beltrão, Luciana Amorim</creatorcontrib><creatorcontrib>Spritzer, Poli Mara</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Publicly Available Content Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>BMC pediatrics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Kopacek, Cristiane</au><au>de Castro, Simone Martins</au><au>Prado, Mayara Jorgens</au><au>da Silva, Claudia Maria Dornelles</au><au>Beltrão, Luciana Amorim</au><au>Spritzer, Poli Mara</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Neonatal screening for congenital adrenal hyperplasia in Southern Brazil: a population based study with 108,409 infants</atitle><jtitle>BMC pediatrics</jtitle><addtitle>BMC Pediatr</addtitle><date>2017-01-17</date><risdate>2017</risdate><volume>17</volume><issue>1</issue><spage>22</spage><epage>22</epage><pages>22-22</pages><artnum>22</artnum><issn>1471-2431</issn><eissn>1471-2431</eissn><abstract>Congenital adrenal hyperplasia (CAH) is an autosomal recessive disorder associated with inborn errors of steroid metabolism. 21-hydroxylase enzyme deficiency occurs in 90 to 95% of all cases of CAH, with accumulation of 17 hydroxyprogesterone (17-OHP). Early diagnosis of CAH based on newborn screening is possible before the development of symptoms and allows proper treatment, correct sex assignment, and reduced mortality rates. This study describes the results obtained in the first year of a public CAH screening program in the state of Rio Grande do Sul, Brazil.
We reviewed the screening database in search of babies with suspected CAH, that is, altered birth-weight adjusted 17-OHP values at screening. The following data were analyzed for this population: screening 17-OHP values, retest 17-OHP values, serum 17-OHP values for those with confirmed CAH on retest, maternal and newborn data, and family history of CAH. For the screening program, 17-OHP levels are determined on dried blood spots obtained in filter paper with GSP solid phase time-resolved immunofluorescence.
Of 108,409 newborns screened, eight were diagnosed with CAH (four males, four females). The incidence of CAH in the state was 1:13,551. Six cases were identified as classic salt-wasting CAH and two were cases of virilizing CAH. The positive predictive value (PPV) of the initial screening (before diagnostic confirmation) was 1.6%. The overall rate of false positive results was 0.47%. The number of false positive results was higher among newborns with birth weight < 2000 g.
The present results support the need for CAH screening by the public health care system in the state, and show that the strategy adopted is adequate. PPV and false positive results were similar to those reported for other states of Brazil with similar ethnic backgrounds.</abstract><cop>England</cop><pub>BioMed Central Ltd</pub><pmid>28095810</pmid><doi>10.1186/s12887-016-0772-x</doi><tpages>1</tpages><oa>free_for_read</oa></addata></record> |
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subjects | 17-alpha-Hydroxyprogesterone - blood Adrenal Hyperplasia, Congenital - blood Adrenal Hyperplasia, Congenital - diagnosis Adrenal Hyperplasia, Congenital - epidemiology Adrenogenital syndrome Analysis Babies Biomarkers - blood Birth weight Brazil - epidemiology Diagnosis Early Diagnosis False Positive Reactions Female Health aspects Humans Hyperplasia Incidence Infant, Newborn Infants (Newborn) Male Medical screening Neonatal Screening - methods Newborn babies Pilot projects Population-based studies Predictive Value of Tests Retrospective Studies Steroids |
title | Neonatal screening for congenital adrenal hyperplasia in Southern Brazil: a population based study with 108,409 infants |
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