Work

Longitudinal Outcomes of a Multimodal Treatment Approach Including Mandibular Distraction Osteogenesis and Continuous Positive Airway Pressure for Pierre Robin Sequence

Year: 2020

Type: article

Abstract: PURPOSE: Mandibular distraction osteogenesis (MDO) and continuous positive airway pressure (CPAP) may effectively treat more and less severe forms of tongue-based airway obstruction in Pierre Robin Sequence, respectively. This study aims to demonstrate safety and longitudinal outcomes for these 2 approaches in accordingly selected patient populations. METHODS: Patients with nonsyndromic Pierre Robin Sequence treated with MDO or CPAP during 2009–2019 were reviewed. Patients who had mild micrognathia, mild glossoptosis on microlaryngoscopy and bronchoscopy (MLB), and obstructive apnea-hypopnea (OAHI) of <20 per hour without significant oxygen desaturations were given the option of CPAP-only treatment; patients with more severe disease generally underwent MDO. Study subjects received baseline and follow-up polysomnography (PSG) examinations. Linear mixed-effect models were used to assess longitudinal predictors of tongue-based airway obstruction, including baseline PSG, age, cleft palate (CP) status, MDO status, and presence of other airway anomalies. RESULTS: One hundred thirty-four patients were treated during the study period: 56 who underwent MDO and 23 who received CPAP-only treatment met inclusion criteria and had 147 and 78 follow-up PSGs, respectively. Median ages at first PSG were 24 days (Q1–Q3 = 9–98 days) and 5 months (1–70 months) for MDO and CPAP-only groups, respectively (P = 0.009). Baseline OAHI was significantly higher in the MDO group (x = 32.9 [20.1–49.1]) compared with CPAP-only group (x = 16.9 [7.4–25.9]; P = 0.002). Baseline maximum ETCO2, SpO2 nadir, mean SpO2, and sleep efficiency did not significantly differ between the 2 groups (P > 0.2). Follow-up OAHI was significantly greater in the MDO compared with the CPAP-only group (Δlog1P = 0.34 [0.03–0.65]; P = 0.031). In the MDO group, patients with repaired and unrepaired CP had significantly higher OAHI after MDO compared with non-CP patients (Δlog1p = 3.22[0.87–5.57], adj-P = 0.008; Δlog1p = 1.42 [0.06–2.79], adj-P = 0.042, respectively). Presence of other airway anomalies was also linked with higher longitudinal OAHI in MDO patients (Δlog1P = 0.48 [0.14–0.81]; adj-P = 0.006). Older age did not lower OAHI longitudinally (adj-P > 0.05). For CPAP-only patients, no longitudinal predictors of OAHI severity were significant (P > 0.05). Follow-up maxETCO2 was not significantly different between MDO and CPAP-only patients (P = 0.217). Patients with higher baseline maxETCO2 demonstrated greater decreases in maxETCO2 after MDO compared with those with lower baseline values (Δ = −0.39 [−0.59, −0.18]; adj-P < 0.001). Patients with repaired CP had higher post-MDO maxETCO2 than non-cleft patients (Δ = 3.77 [0.04–7.54]; adj-P = 0.049). Follow-up SpO2 nadir was not significantly different between MDO and CPAP-only patients (P = 0.132). Older age and other airway anomalies were, respectively, associated with higher and lower SpO2 nadir (adj-P = 0.044; 0.041). Patients with repaired and unrepaired CP had higher SpO2 nadir compared with non-cleft patients (adj-P = 0.002; 0.001). MDO status did not influence SpO2 nadir longitudinally (adj-P = 0.808). Similar trends were observed for mean SpO2 over time. Longitudinally, sleep efficiency did not differ between MDO and CPAP-only patients (P = 0.94) and had no significant predictors (adj-P > 0.05). CONCLUSIONS: After controlling for potential confounders, MDO effectively improves OAHI and maxETCO2 longitudinally in patients with Pierre Robin sequence; this benefit is more pronounced in noncleft than in CP patients. Our analysis does not detect any significant predictor of PSG outcomes over time in CPAP-only patients. Additional analyses are warranted to further assess longitudinal outcomes of CPAP-only treatment for the selected group of patients with milder disease.

Source: Plastic & Reconstructive Surgery Global Open

DOI: 10.1097/01.gox.0000720636.78508.ab