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Airway management in infants with Robin sequence in the United Kingdom and Ireland: A prospective population-based study

Wright, Marie; Knowles, Rachel L; Cortina-Borja, Mario; Javadpour, Sheila; Mehendale, Felicity V; Urquhart, Don S; (2024) Airway management in infants with Robin sequence in the United Kingdom and Ireland: A prospective population-based study. Pediatric Pulmonology , 59 (11) pp. 2839-2849. 10.1002/ppul.27140. Green open access

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Abstract

Objective: There is currently no consensus about managing upper airway obstruction (UAO) in infants with Robin sequence (RS), in terms of treatment efficacy or clinical outcomes. This study describes UAO management in UK/Ireland, and explores relationships between patient characteristics, UAO management, and clinical outcomes in the first 2 years of life. // Methods: Active surveillance of RS throughout UK/Ireland via the British Paediatric Surveillance Unit and nationally commissioned cleft services. Clinical data were collected at initial notification and 12-month follow-up. // Results: 173 infants with RS were identified, of which 47% had additional congenital anomalies or an underlying syndrome (non-isolated RS). Two-thirds (n = 119) required an airway intervention other than prone positioning: non-surgical in 84% and surgical (tracheostomy) in 16%. Nasopharyngeal airway (NPA) was the most common intervention, used in 83% (n = 99) for median 90 days (IQR 136). Surgical UAO management was associated with prolonged hospital admission, higher prevalence of neurodevelopmental delay (NDD), lower weight-for-age z-scores, and delayed oral feeding. These findings were not attributable to a higher prevalence of non-isolated RS in this group. Although more commonly associated with non-isolated RS, growth faltering was also identified in 48%, and NDD in 18%, of cases of isolated RS. // Conclusions: In UK/Ireland, most infants with RS are managed with NPA, and tracheostomy is reserved for refractory severe UAO. Clinical outcomes and duration of use indicate that NPA is a safe and feasible first-line approach to UAO. Longitudinal assessment of neurodevelopment and growth is imperative, including in children with isolated RS. Current variations in practice reinforce the need for evidence-based treatment guidelines.

Type: Article
Title: Airway management in infants with Robin sequence in the United Kingdom and Ireland: A prospective population-based study
Location: United States
Open access status: An open access version is available from UCL Discovery
DOI: 10.1002/ppul.27140
Publisher version: https://doi.org/10.1002/ppul.27140
Language: English
Additional information: Copyright © 2024 The Author(s). Pediatric Pulmonology published by Wiley Periodicals LLC. This is an open access article under the terms of the Creative Commons Attribution License, https://creativecommons.org/licenses/by/4.0/, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
Keywords: Airway obstruction, congenital abnormalities, management strategies, outcomes, Robin sequence
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.ucl.ac.uk/id/eprint/10206553
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