Characterizing Differences in Polysomnography Data for Children With Robin Sequence Undergoing Conservative and Surgical Management.

Abstract

To perform a meta-analysis of polysomnography (PSG) findings in infants with Robin sequence (RS). CINAHL, Cochrane Library, PubMed, and Scopus. Studies describing PSG results in infants with RS before and/or after intervention were included. Meta-analysis of mean difference (Δ), proportions (%), and continuous measures with 95% confidence interval (CI) were calculated using random and fixed effects models. In total, 45 studies including 1881 infants were analyzed. The mean age at PSG was 24.89 (range 0-267) days, and 27.92% (CI 21.7-34.7) had an associated syndrome, with Stickler syndrome being most common (11.08%, CI 9.3-13.1). At baseline, 79.88% (CI 59.1-94.5) were diagnosed with severe obstructive sleep apnea. Most patients underwent a mandibular distraction osteogenesis (MDO, 66%), followed by conservative management (14%) or tongue-lip adhesion (TLA, 11%). Baseline apnea-hypopnea index (AHI) was highest in the TLA group (39.05/h, CI 8.4-69.7), compared to the MDO (31.48/h, CI 27.5-35.5) and conservative treatment groups (19.92/h, CI 15.6-24.2). There was an improvement in AHI following surgery by 24.52/h (CI 21.4-27.7) (P < .001) after MDO and by 25.38/h (CI 1.4-49.3) (P = .04) following TLA. Additionally, SpO₂ nadir increased by 11.49% (CI 9.7-13.3) following MDO. TLA had slightly higher rates of postoperative tracheostomy (5.33%, CI 2.9-9.4) compared to MDO (3.8%, CI 1.8-6.5). Infants with RS with an AHI approaching 30 tended to undergo surgical intervention. PSG findings improved significantly following MDO and TLA, with few requiring further surgeries including tracheostomy. Future research should explore integrating PSG results with clinical data to inform an algorithm for management.