Objective measurements for upper airway obstruction in infants with Robin sequence: what are we measuring? A systematic review

Robrecht J.H. Logjes, Joanna E. MacLean, Noor W. de Cort, Christian F. Poets, Véronique Abadie, Koen F.M. Joosten, Cory M. Resnick, Ivy K. Trindade-Suedam, Carlton J. Zdanski, Christopher R. Forrest, Frea H. Kruisinga, Roberto L. Flores, Kelly N. Evans, Corstiaan C. Breugem*

*Corresponding author for this work

Research output: Contribution to journalReview articleAcademicpeer-review

13 Citations (Scopus)


Study Objectives: Identifying optimal treatment for infants with Robin sequence (RS) is challenging due to substantial variability in the presentation of upper airway obstruction (UAO) in this population.Objective assessments of UAOand treatments are not standardized. A systematic review of objectivemeasures ofUAO was conducted as a step toward evidence-based clinical decision-making for RS. Methods: A literature search was performed in the PubMed and Embase databases (1990-2020) following PRISMA guidelines. Articles reporting on RS and UAO treatment were included if the following objective measures were studied: Oximetry, polysomnography, and blood gas. Quality was appraised by the methodological index for nonrandomized studies (range: 0-24). Results: A total of 91 articles met the inclusion criteria. The mean methodological index for nonrandomized studies score was 7.1 (range: 3-14). Polysomnography was most frequently used (76%) followed by oximetry (20%) and blood gas (11%). Sleep position of the infantwas reported in 35%of studies, with supine position most frequently, and monitoring time in 42%, including overnight recordings, in more than half. Of 71 studies that evaluated UAO interventions, the majority used polysomnography (90%), of which 61% did not specify the polysomnography technique. Reported polysomnography metrics included oxygen saturation (61%), apnea-hypopnea index (52%), carbon dioxide levels (31%), obstructive apnea-hypopnea index (27%), and oxygen desaturation index (16%). Only 42 studies reported indications for UAO intervention, with oximetry and polysomnography thresholds used equally (both 40%). In total, 34 distinct indications for treatment were identified. Conclusions: This systematic review demonstrates a lack of standardization, interpretation, and reporting of assessment and treatment indications for UAO in RS. An international, multidisciplinary consensus protocol is needed to guide clinicians on optimal UAO assessment in RS.

Original languageEnglish
Pages (from-to)1717-1729
Number of pages13
JournalJournal of Clinical Sleep Medicine
Issue number8
Early online date1 Aug 2021
Publication statusPublished - 2021

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© 2021 American Academy of Sleep Medicine.


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