Wright, M;
Cortina-Borja, M;
Knowles, R;
Urquhart, DS;
(2023)
Global birth prevalence of Robin sequence in live-born infants: a systematic review and meta-analysis.
European Respiratory Review
, 32
(170)
, Article 230133. 10.1183/16000617.0133-2023.
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Abstract
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 versus non-registry data). The protocol was prospectively registered with PROSPERO. Databases were searched using keywords and subject terms for “Robin sequence”, “epidemiology”, “incidence” and “birth prevalence”. Meta-analysis was performed fitting random effects models with arcsine transformation. From 34 eligible studies (n=2722 RS cases), pooled birth prevalence was 9.5 per 100 000 live births (95% CI 7.1–12.1) 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. 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.
| Type: | Article |
|---|---|
| Title: | Global birth prevalence of Robin sequence in live-born infants: a systematic review and meta-analysis |
| Location: | England |
| Open access status: | An open access version is available from UCL Discovery |
| DOI: | 10.1183/16000617.0133-2023 |
| Publisher version: | https://doi.org/10.1183/16000617.0133-2023 |
| Language: | English |
| Additional information: | © The authors 2023 This version is distributed under the terms of the Creative Commons Attribution Non-Commercial Licence 4.0. For commercial reproduction rights and permissions contact permissions@ersnet.org |
| Keywords: | Infant, Humans, Pierre Robin Syndrome, Prevalence, Incidence, Registries, Consensus |
| UCL classification: | UCL UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Population, Policy and Practice Dept |
| URI: | https://discovery-pp.ucl.ac.uk/id/eprint/10184374 |
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