{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T04:33:11Z","timestamp":1777523591456,"version":"3.51.4"},"reference-count":44,"publisher":"SAGE Publications","issue":"12","license":[{"start":{"date-parts":[[2024,10,9]],"date-time":"2024-10-09T00:00:00Z","timestamp":1728432000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["The Cleft Palate Craniofacial Journal"],"published-print":{"date-parts":[[2025,12]]},"abstract":"<jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To determine whether children born with a cleft palate\u2009\u00b1\u2009lip (CP\u2009\u00b1\u2009L) and additional congenital differences (ACDs \u2013 including \u2018Congenital malformations and deformations\u2019 as coded in ICD-10), are less likely to meet the three national speech outcome standards at age five compared to children with CP\u2009\u00b1\u2009L and no ACDs.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>An observational study, utilizing national data from the UK Cleft Registry and Audit NEtwork (CRANE) Database linked to national administrative data of hospital admissions.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Setting<\/jats:title>\n                    <jats:p>National Health Service, England.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Patients<\/jats:title>\n                    <jats:p>2191 children (993 female, 1198 male) with CP\u2009\u00b1\u2009L, born 2006-2012 in England, with complete speech data and consent to data linkage.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Outcome Measure<\/jats:title>\n                    <jats:p>Perceptual speech analysis utilised the Cleft Audit Protocol for Speech \u2013 Augmented (CAPS-A) rating and United Kingdom National Speech Outcome Standards (Speech Standard 1 (SS1), Speech Standard 2a (SS2a) and Speech Standard 3 (SS3)).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Of 2191 children, 759 (35%) had at least one ACD. Presence of one ACD did not significantly impact speech outcomes but two or more ACDs reduced the odds of achieving all three speech standards: SS1 aOR 0.602 (CI 0.45-0.82,\n                      <jats:italic toggle=\"yes\">P<\/jats:italic>\n                      \u2009=\u2009.002), SS2a aOR 0.563 (CI 0.41-0.77,\n                      <jats:italic toggle=\"yes\">P<\/jats:italic>\n                      \u2009=\u2009.001), SS3 aOR 0.606 (0.43-0.84,\n                      <jats:italic toggle=\"yes\">P<\/jats:italic>\n                      \u2009=\u2009.003). When exploring ACDs by ICD-10 groupings, congenital malformations of the \u2018Eye, ear, face and neck\u2019, \u2018Circulatory system\u2019, \u2018Digestive system\u2019, \u2018Musculoskeletal system\u2019 and \u2018Other congenital malformations\u2019 reduced a child's odds of achieving the speech standards.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>ACDs, in the absence of a known syndrome, have a significant impact on speech outcome at age five. Incorporating these factors into risk-adjustment models for service level outcome reporting is recommended.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/10556656241287759","type":"journal-article","created":{"date-parts":[[2024,10,9]],"date-time":"2024-10-09T21:16:19Z","timestamp":1728508579000},"page":"2155-2166","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":3,"title":["Investigating the Impact of Additional Congenital Malformations on Speech Outcomes at age Five in Children with a Cleft Palate"],"prefix":"10.1177","volume":"62","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5867-2625","authenticated-orcid":false,"given":"Sophie","family":"Butterworth","sequence":"first","affiliation":[{"name":"The Royal College of Surgeons of England"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2385-3172","authenticated-orcid":false,"given":"Kate J.","family":"Fitzsimons","sequence":"additional","affiliation":[{"name":"The Royal College of Surgeons of England"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lorraine","family":"Britton","sequence":"additional","affiliation":[{"name":"Nottingham University Hospital NHS Trust"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0902-0564","authenticated-orcid":false,"given":"Stephanie","family":"Van Eeden","sequence":"additional","affiliation":[{"name":"School of Education, Communication and Language Sciences, Newcastle University"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jibby","family":"Medina","sequence":"additional","affiliation":[{"name":"The Royal College of Surgeons of England"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jan","family":"van der Meulen","sequence":"additional","affiliation":[{"name":"London School of Hygiene and Tropical Medicine, London, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Craig J.H.","family":"Russell","sequence":"additional","affiliation":[{"name":"Royal Hospital for Children"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2024,10,9]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1159\/000337464"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(82)80261-8"},{"key":"e_1_3_3_4_2","volume-title":"International classification of diseases tenth revision (ICD-10)","year":"2019","unstructured":"International classification of diseases tenth revision (ICD-10). 6th ed. 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