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Characterizing Differences in Polysomnography Data for Children With Robin Sequence Undergoing Conservative and
Angelica M Walker1, Thomas Gao1, Shaun A Nguyen1
1Department of Otolaryngology-Head and Neck Surgery, Medical University of South Carolina, Charleston, South Carolina, USA.
Insights
Infants with Robin sequence (RS) often have severe obstructive sleep apnea. Surgical interventions like mandibular distraction osteogenesis (MDO) and tongue-lip adhesion (TLA) significantly improve sleep apnea findings.
Area of Science:
- Pediatric Sleep Medicine
- Craniofacial Surgery
- Genetics
Background:
- Robin sequence (RS) is a congenital condition characterized by micrognathia, glossoptosis, and airway obstruction.
- Obstructive sleep apnea (OSA) is a common and serious comorbidity in infants with RS, impacting their health and development.
Purpose of the Study:
- To conduct a meta-analysis of polysomnography (PSG) findings in infants diagnosed with Robin sequence (RS).
- To evaluate the effectiveness of different interventions for sleep-disordered breathing in this population.
Main Methods:
- A systematic literature search was performed across CINAHL, Cochrane Library, PubMed, and Scopus.
- Meta-analysis of mean differences, proportions, and continuous measures with 95% confidence intervals was conducted using random and fixed effects models for studies reporting PSG results before and after intervention.
Main Results:
- Analysis of 45 studies (1881 infants) revealed that 79.88% of infants with RS presented with severe obstructive sleep apnea at baseline.
- Mandibular distraction osteogenesis (MDO) and tongue-lip adhesion (TLA) interventions led to significant improvements in the apnea-hypopnea index (AHI) and SpO2 nadir.
- While both MDO and TLA showed efficacy, TLA had slightly higher rates of postoperative tracheostomy.
Conclusions:
- Infants with RS and an AHI nearing 30 commonly undergo surgical intervention.
- PSG findings demonstrate significant improvement post-MDO and TLA, with a low need for further surgical procedures like tracheostomy.
- Future research should focus on integrating PSG data with clinical information to develop management algorithms for RS-related sleep apnea.
Objective:
To perform a meta-analysis of polysomnography (PSG) findings in infants with Robin sequence (RS).
Data Sources:
CINAHL, Cochrane Library, PubMed, and Scopus.
Review Methods:
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.
Results:
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).
Conclusion:
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.
