{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2023,9,13]],"date-time":"2023-09-13T05:40:06Z","timestamp":1694583606615},"reference-count":14,"publisher":"Wiley","issue":"11","license":[{"start":{"date-parts":[[2009,9,23]],"date-time":"2009-09-23T00:00:00Z","timestamp":1253664000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2009,11]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>BACKGROUND<\/jats:title><jats:p>Hirschsprung's disease (HSCR) is a potentially fatal congenital intestinal obstruction syndrome. For birth years 1996\u20132002, reported HSCR prevalence in Alaska was approximately six times higher than the national estimate of two per 10,000 live births. In 2008 we completed a case verification study of HSCR reported to the Alaska Birth Defects Registry (ABDR) to verify prevalence and evaluate sensitivity and predictive value of HSCR surveillance.<\/jats:p><\/jats:sec><jats:sec><jats:title>METHODS<\/jats:title><jats:p>We abstracted medical records for children reported with HSCR who were born in 1996\u20132007 and matched to an Alaska birth certificate. We categorized reports as confirmed, ruled out, or inconclusive. ABDR sensitivity was validated by cross\u2010referencing hospital discharge data with confirmed HSCR cases.<\/jats:p><\/jats:sec><jats:sec><jats:title>RESULTS<\/jats:title><jats:p>Overall sensitivity of the ABDR was 94.4%, and positive predictive value (PPV) was 31.1%. Following case verification, HSCR prevalence was three per 10,000 live births statewide and 5.6 and 2.1 per 10,000 live births for Alaska Natives and non\u2010Natives, respectively. Positive predictive value was twice as high for the Alaska Native population as the non\u2010Native population.<\/jats:p><\/jats:sec><jats:sec><jats:title>CONCLUSIONS<\/jats:title><jats:p>Multiple\u2010source passive surveillance methodology resulted in high sensitivity for HSCR ascertainment; however, case verification was required to rule out false\u2010positive reports, which obscured the true distribution of Alaska Native versus non\u2010Native HSCR cases. Birth Defects Research (Part A) 2009. \u00a9 2009 Wiley\u2010Liss, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/bdra.20628","type":"journal-article","created":{"date-parts":[[2009,9,23]],"date-time":"2009-09-23T13:21:44Z","timestamp":1253712104000},"page":"914-919","source":"Crossref","is-referenced-by-count":6,"title":["False positive reporting of Hirschsprung's disease in Alaska: An evaluation of Hirschsprung's disease surveillance, birth years 1996\u20132007"],"prefix":"10.1002","volume":"85","author":[{"given":"Janine","family":"Schoellhorn","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sandra","family":"Collins","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2009,9,23]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.38.11.729"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/pds.1534"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1093\/aje\/155.9.866"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20212"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1520-6661(199707\/08)6:4<230::AID-MFM9>3.0.CO;2-K"},{"key":"e_1_2_6_7_1","volume-title":"Using Medicaid data to track asthma in New England: a status report","author":"Lichter E","year":"2004"},{"key":"e_1_2_6_8_1","volume-title":"North Carolina birth defects monitoring program","author":"Morgan A","year":"2000"},{"key":"e_1_2_6_9_1","first-page":"906","article-title":"State birth defects surveillance program directory","volume":"82","author":"National Birth Defects Prevention Network","year":"2008","journal-title":"Birth Defects Res A Clin Mol Teratol"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.2105\/AJPH.85.12.1706"},{"key":"e_1_2_6_11_1","volume-title":"Alaska maternal and child health databook 2005: birth defects edition","author":"Schoellhorn KJ","year":"2006"},{"key":"e_1_2_6_12_1","volume-title":"The value of hospital discharge databases","author":"Schoenman JA","year":"2005"},{"key":"e_1_2_6_13_1","volume-title":"Improvement of state Medicaid data quality and use in eliminating racial and ethnic disparities","author":"Shaw\u2010Taylor Y","year":"2001"},{"key":"e_1_2_6_14_1","volume-title":"Conditions reportable to public health","author":"State of Alaska","year":"2008"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.2105\/AJPH.86.5.731"}],"container-title":["Birth Defects Research Part A: Clinical and Molecular Teratology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fbdra.20628","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/bdra.20628","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,12]],"date-time":"2023-09-12T10:14:14Z","timestamp":1694513654000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/bdra.20628"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2009,9,23]]},"references-count":14,"journal-issue":{"issue":"11","published-print":{"date-parts":[[2009,11]]}},"alternative-id":["10.1002\/bdra.20628"],"URL":"https:\/\/doi.org\/10.1002\/bdra.20628","archive":["Portico"],"relation":{},"ISSN":["1542-0752","1542-0760"],"issn-type":[{"value":"1542-0752","type":"print"},{"value":"1542-0760","type":"electronic"}],"subject":[],"published":{"date-parts":[[2009,9,23]]}}}