Global birth prevalence of Robin sequence in live-born infants: a systematic review and meta-analysis.
Marie WrightMario Cortina-BorjaRachel KnowlesDonald S UrquhartPublished in: European respiratory review : an official journal of the European Respiratory Society (2023)
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.
Keyphrases
- risk factors
- healthcare
- case control
- gestational age
- systematic review
- single cell
- public health
- randomized controlled trial
- emergency department
- mental health
- big data
- pregnant women
- electronic health record
- risk assessment
- climate change
- meta analyses
- clinical practice
- preterm birth
- study protocol
- pregnancy outcomes
- low birth weight