{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,31]],"date-time":"2026-08-31T11:11:02Z","timestamp":1788174662261,"version":"build-2803163510"},"reference-count":60,"publisher":"European Respiratory Society (ERS)","issue":"170","license":[{"start":{"date-parts":[[2023,12,6]],"date-time":"2023-12-06T00:00:00Z","timestamp":1701820800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["publications.ersnet.org"],"crossmark-restriction":true},"short-container-title":["Eur Respir Rev"],"accepted":{"date-parts":[[2023,10,16]]},"published-print":{"date-parts":[[2023,12,31]]},"abstract":"<jats:p>\n                    Robin sequence (RS), a congenital disorder of jaw maldevelopment and glossoptosis, poses a substantial healthcare burden and has long-term health implications if airway obstruction is suboptimally treated. This study describes the global birth prevalence of RS and investigates whether prevalence estimates differ by geographical location, ethnicity or study data source (registry\n                    <jats:italic>versus<\/jats:italic>\n                    non-registry data). The protocol was prospectively registered with PROSPERO.\n                  <\/jats:p>\n                  <jats:p>Databases were searched using keywords and subject terms for \u201cRobin sequence\u201d, \u201cepidemiology\u201d, \u201cincidence\u201d and \u201cbirth prevalence\u201d. Meta-analysis was performed fitting random effects models with arcsine transformation.<\/jats:p>\n                  <jats:p>From 34 eligible studies (n=2722 RS cases), pooled birth prevalence was 9.5 per 100\u2009000 live births (95% CI 7.1\u201312.1) with statistical heterogeneity. One third of studies provided a case definition for RS and numerous definitions were used. A total of 22 countries were represented, predominantly from European populations (53% of studies). There was a trend towards higher birth prevalence in European populations and lower prevalence from registry-based studies. Only two studies reported ethnicity.<\/jats:p>\n                  <jats:p>This study indicates that RS occurs globally. To investigate geographical differences in prevalence, additional studies from non-European populations and reporting of ethnicity are needed. Heterogeneity of estimates may be due to variable diagnostic criteria and ascertainment methods. Recently published consensus diagnostic criteria may reduce heterogeneity among future studies.<\/jats:p>","DOI":"10.1183\/16000617.0133-2023","type":"journal-article","created":{"date-parts":[[2023,12,6]],"date-time":"2023-12-06T04:45:14Z","timestamp":1701837914000},"page":"230133","update-policy":"https:\/\/doi.org\/10.1183\/ers-crossmark-policy","source":"Crossref","is-referenced-by-count":20,"title":["Global birth prevalence of Robin sequence in live-born infants: a systematic review and meta-analysis"],"prefix":"10.1183","volume":"32","author":[{"given":"Marie","family":"Wright","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mario","family":"Cortina-Borja","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel","family":"Knowles","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9048-9692","authenticated-orcid":false,"given":"Don S.","family":"Urquhart","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"81","published-online":{"date-parts":[[2023,12,6]]},"reference":[{"key":"2024102404163870000_32.170.230133.1","doi-asserted-by":"crossref","first-page":"894","DOI":"10.1001\/jamapediatrics.2016.0796","article-title":"Best practices for the diagnosis and evaluation of infants with Robin sequence: a clinical consensus report","volume":"170","author":"Breugem","year":"2016","journal-title":"JAMA Pediatr"},{"key":"2024102404163870000_32.170.230133.2","doi-asserted-by":"crossref","first-page":"530","DOI":"10.1111\/ppe.12776","article-title":"Epidemiology of Pierre\u2013Robin sequence in Europe: a population-based EUROCAT study","volume":"35","author":"Santoro","year":"2021","journal-title":"Paediatr Perinat Epidemiol"},{"key":"2024102404163870000_32.170.230133.3","doi-asserted-by":"publisher","DOI":"10.1136\/archdischild-2014-306472"},{"key":"2024102404163870000_32.170.230133.4","doi-asserted-by":"crossref","first-page":"1972","DOI":"10.1002\/ajmg.a.37150","article-title":"Birth prevalence of Robin sequence in the Netherlands from 2000\u20132010: a retrospective population-based study in a large Dutch cohort and review of the literature","volume":"167A","author":"Paes","year":"2015","journal-title":"Am J Med Genet A"},{"key":"2024102404163870000_32.170.230133.5","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.n71"},{"key":"2024102404163870000_32.170.230133.6","doi-asserted-by":"publisher","DOI":"10.1001\/jama.283.15.2008"},{"key":"2024102404163870000_32.170.230133.7","doi-asserted-by":"publisher","DOI":"10.1016\/j.jbi.2008.08.010"},{"key":"2024102404163870000_32.170.230133.8","doi-asserted-by":"publisher","DOI":"10.1097\/XEB.0000000000000054"},{"key":"2024102404163870000_32.170.230133.9","doi-asserted-by":"publisher","DOI":"10.1136\/jech-2013-203104"},{"key":"2024102404163870000_32.170.230133.10","doi-asserted-by":"publisher","DOI":"10.1097\/XEB.0000000000000065"},{"key":"2024102404163870000_32.170.230133.11","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.327.7414.557"},{"key":"2024102404163870000_32.170.230133.12","doi-asserted-by":"publisher","DOI":"10.1016\/S0895-4356(01)00377-8"},{"key":"2024102404163870000_32.170.230133.13","doi-asserted-by":"crossref","first-page":"153","DOI":"10.1136\/ebmental-2019-300117","article-title":"How to perform a meta-analysis with R: a practical tutorial","volume":"22","author":"Balduzzi","year":"2019","journal-title":"Evid Based Ment Health"},{"key":"2024102404163870000_32.170.230133.14","doi-asserted-by":"crossref","first-page":"1","DOI":"10.18637\/jss.v036.i03","article-title":"Conducting meta-analyses in R with the metafor package","volume":"36","author":"Viechtbauer","year":"2010","journal-title":"J Stat Soft"},{"key":"2024102404163870000_32.170.230133.15","doi-asserted-by":"crossref","first-page":"1","DOI":"10.18637\/jss.v067.i01","article-title":"Fitting linear mixed-effects models using lme4","volume":"67","author":"Bates","year":"2015","journal-title":"J Stat Soft"},{"key":"2024102404163870000_32.170.230133.16","doi-asserted-by":"crossref","first-page":"1259","DOI":"10.1177\/1055665620929247","article-title":"Prevalence and clinical case series of syndromic and nonsyndromic cleft lip and palate in a Saudi Arabian neonatal population","volume":"57","author":"Alyami","year":"2020","journal-title":"Cleft Palate Craniofac J"},{"key":"2024102404163870000_32.170.230133.17","doi-asserted-by":"publisher","DOI":"10.1093\/ortho\/26.2.121"},{"key":"2024102404163870000_32.170.230133.18","doi-asserted-by":"publisher","DOI":"10.1093\/ejo\/cjq117"},{"key":"2024102404163870000_32.170.230133.19","first-page":"256","article-title":"Major congenital malformations presenting in the first 24\u2005h of life in 3865 consecutive births in south of Beirut. 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