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

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 UAO and treatments are not standardized. A systematic review of objective measures of UAO was conducted as step towards evidence based clinical decision making for RS. Methods A literature search was performed in 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 methodological index for non-randomized studies (MINORS, range:0-24). Results A total of 91 articles met inclusion criteria. Mean MINORS-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 infant was 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.