{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,3]],"date-time":"2026-05-03T01:39:12Z","timestamp":1777772352795,"version":"3.51.4"},"reference-count":18,"publisher":"Wiley","issue":"11","license":[{"start":{"date-parts":[[2025,11,18]],"date-time":"2025-11-18T00:00:00Z","timestamp":1763424000000},"content-version":"vor","delay-in-days":17,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2025,11,1]],"date-time":"2025-11-01T00:00:00Z","timestamp":1761955200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100000082","name":"Cystic Fibrosis Canada","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000082","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Pulmonology"],"published-print":{"date-parts":[[2025,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Early diagnosis of cystic fibrosis (CF) through newborn screening (NBS) programs has improved health outcomes in people with CF (pwCF). NBS programs can vary in specific protocols and genetic variants tested, which may not perform equitably for all infants. The objective of this study was to summarize the Canadian CF NBS programs to understand if there are any gaps that may drive inequities.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Details about each of the Canadian CF NBS programs were gathered by collating publicly available information and consulting directly with each program. The Canadian CF Registry (CCFR) was used to identify Canadians with CF in 2022, to estimate the proportion of individuals that would have been identified by each NBS program in Canada.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      All jurisdictions in Canada include CF in their NBS programs, which follow a similar multistep process: (1) evaluation of immunoreactive trypsinogen (IRT), (2) genetic testing of a predefined set of variants. Most jurisdictions analyzed IRT locally, whereas genetic testing was centralized to five programs. Applying the current NBS\n                      <jats:italic>CFTR<\/jats:italic>\n                      variant panels from each program to the 4445 individuals in the CCFR identified over 96% of the Canadian CF population. All variant screening panels were more likely to identify pwCF who were: (1) born before 2018, (2) diagnosed as children, and (3) described as White.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interpretation<\/jats:title>\n                    <jats:p>Canadian NBS panels would have captured over 96% of all people in the CCFR; however, they would fail to identify 12%\u201320% of non\u2010White individuals. As Canada's population becomes more diverse, updates to NBS programs may be needed to ensure inequities in screening and diagnosis do not further widen.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ppul.71386","type":"journal-article","created":{"date-parts":[[2025,11,19]],"date-time":"2025-11-19T07:04:59Z","timestamp":1763535899000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["A Review of Newborn Screening Programs for Cystic Fibrosis: Are Current Protocols Appropriate for Canada's Diverse Population?"],"prefix":"10.1002","volume":"60","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2890-8451","authenticated-orcid":false,"given":"Stephanie Y.","family":"Cheng","sequence":"first","affiliation":[{"name":"Cystic Fibrosis Canada Toronto Ontario Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-9797-8492","authenticated-orcid":false,"given":"Berke","family":"Sahin","sequence":"additional","affiliation":[{"name":"Dalhousie University Halifax Nova Scotia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Noma","family":"Abdulrahem","sequence":"additional","affiliation":[{"name":"Cystic Fibrosis Canada Toronto Ontario Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paul D. W.","family":"Eckford","sequence":"additional","affiliation":[{"name":"Cystic Fibrosis Canada Toronto Ontario Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zofia","family":"Zysman\u2010Colman","sequence":"additional","affiliation":[{"name":"Montreal Children's Hospital Montreal Quebec Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mark","family":"Chilvers","sequence":"additional","affiliation":[{"name":"BC Children's Hospital Vancouver British Columbia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4529-5653","authenticated-orcid":false,"given":"Anne L.","family":"Stephenson","sequence":"additional","affiliation":[{"name":"St. Michael's Hospital Toronto Ontario Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jocelyn","family":"Arpin","sequence":"additional","affiliation":[{"name":"Winnipeg Children's Hospital Winnipeg Manitoba Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christine","family":"Donnelly","sequence":"additional","affiliation":[{"name":"IWK Health Centre Halifax Nova Scotia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Karen","family":"Doyle","sequence":"additional","affiliation":[{"name":"Janeway Children's Health Centre, St. John's, Newfoundland  Canada"},{"name":"Health Sciences Centre, St. John's, Newfoundland  Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sara","family":"Fernandez","sequence":"additional","affiliation":[{"name":"Newborn Screening Ontario, Children's Hospital of Eastern Ontario Ottawa Ontario Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Corey","family":"Filiaggi","sequence":"additional","affiliation":[{"name":"Maritime Newborn Screening, IWK Health Centre Halifax Nova Scotia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zaiping","family":"Liu","sequence":"additional","affiliation":[{"name":"Maritime Newborn Screening, IWK Health Centre Halifax Nova Scotia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Angela","family":"Nowak","sequence":"additional","affiliation":[{"name":"BC Children's Hospital Vancouver British Columbia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mary Jane","family":"Smith","sequence":"additional","affiliation":[{"name":"Janeway Children's Health Centre, St. John's, Newfoundland  Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7931-8051","authenticated-orcid":false,"given":"Sanja","family":"Stanojevic","sequence":"additional","affiliation":[{"name":"Dalhousie University Halifax Nova Scotia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,11,18]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamapediatrics.2022.2674"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.107.1.1"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2016.04.001"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1183\/23120541.00109\u20102017"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2017.01.016"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2016.48"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/ppul.26279"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2022.07.010"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijns11020024"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2024.09.009"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2745"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/ppul.26253"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1164\/rccm.202301\u20100021OC"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2023.03.002"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijns8040058"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2021.03.006"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcf.2021.12.004"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijns9010010"}],"container-title":["Pediatric Pulmonology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ppul.71386","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/ppul.71386","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ppul.71386","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,28]],"date-time":"2026-01-28T08:59:28Z","timestamp":1769590768000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ppul.71386"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,11]]},"references-count":18,"journal-issue":{"issue":"11","published-print":{"date-parts":[[2025,11]]}},"alternative-id":["10.1002\/ppul.71386"],"URL":"https:\/\/doi.org\/10.1002\/ppul.71386","archive":["Portico"],"relation":{},"ISSN":["8755-6863","1099-0496"],"issn-type":[{"value":"8755-6863","type":"print"},{"value":"1099-0496","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,11]]},"assertion":[{"value":"2025-04-21","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-11-04","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-11-18","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e71386"}}