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The Role of Ankyloglossia as a Protective Factor in Management of Infants With Robin Sequence
David O'Neil Danis1, Peter L Steinwald1, Andrew R Scott1,2,3
1Dr. Elie E. Rebeiz Department of Otolaryngology-Head and Neck Surgery, Tufts Medical Center, Boston, Massachusetts, USA.
Insights
Ankyloglossia (tongue-tie) may protect infants with Robin sequence (RS) from breathing problems and the need for airway interventions. This finding suggests a potential protective role for tongue-tie in managing RS complications.
Area of Science:
- Pediatric Surgery
- Craniofacial Anomalies
- Neonatal Care
Background:
- Robin sequence (RS) is a congenital anomaly characterized by micrognathia, glossoptosis, and airway obstruction.
- Infants with RS often experience significant breathing difficulties and require interventions.
- The role of associated conditions, such as ankyloglossia, in modifying RS outcomes is not fully understood.
Purpose of the Study:
- To investigate the potential protective effect of ankyloglossia in infants diagnosed with Robin sequence.
- To determine if the presence of ankyloglossia influences the severity of respiratory compromise and the need for airway interventions in RS patients.
Main Methods:
- Retrospective analysis of a 15-year single-institution chart review of infants with RS.
- Comparison with a larger, population-based analysis using the Kids' Inpatient Database (2016 and 2019).
- Statistical analysis included prevalence rates and odds ratios (ORs) to quantify associations between ankyloglossia and clinical outcomes.
Main Results:
- In the single-institution cohort, ankyloglossia was present in 8.8% of infants with RS and was associated with reduced hypoventilation, fewer airway interventions, and shorter hospital stays.
- The nationwide analysis revealed ankyloglossia in 3.7% of RS births, showing a negative association with respiratory failure (adjusted OR 0.19) and mechanical ventilation need.
- Ankyloglossia was significantly linked to a decreased likelihood of requiring respiratory support and airway interventions in both study cohorts.
Conclusions:
- Ankyloglossia may serve as a protective factor in infants with Robin sequence.
- The presence of ankyloglossia appears to reduce the risk of hypoventilation, respiratory failure, and the need for mechanical ventilation.
- These findings highlight the importance of evaluating for ankyloglossia in infants with RS and may inform management strategies.
Objective:
Robin sequence (RS) is an anomaly of micrognathia, glossoptosis, and airway obstruction. This study aims to assess the role of ankyloglossia as a protective factor in infants with RS.
Study Design:
Single institution and population-based inpatient registry analysis.
Setting:
Academic medical center.
Methods:
A retrospective 15-year single-institution chart review examined medical records of infants with RS at an urban tertiary care center. Data included patient characteristics, comorbidities/complications, airway interventions, and length of stay (LOS). Conclusions from this smaller sample were compared with analyses from a larger, less detailed, population-based inpatient registry using the Kids' Inpatient Database (2016 and 2019). To quantify associations between patient characteristics and interventions and ankyloglossia, prevalence rates and unadjusted and adjusted odds ratios (ORs) were used.
Results:
In the single-institution analysis, five (8.8%) of 57 infants with RS had ankyloglossia. Ankyloglossia was negatively associated with hypoventilation (P = .024), need for airway intervention (P < .001), number of airway interventions (P < .001), and total and NICU LOS (P = .001). In the nationwide analysis, 45 (3.7%) of 1207 weighted hospital births with RS had ankyloglossia. Ankyloglossia was negatively associated with respiratory failure of newborn in both univariate and multivariate analysis (unadjusted OR 0.44, P = .027; adjusted OR 0.19, P = .005), as well as need for mechanical ventilation in univariate analysis (unadjusted OR 0.39, P = .022).
Conclusion:
In infants with RS, ankyloglossia may be protective against hypoventilation, mechanical ventilation, and the need for airway intervention.
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