| Presenter: Melinda Costa, MD, Indiana University
Authors: Melinda Costa (1), Michael Tu (1), Kariuki Murage (2), Robert J. Havlik (1), Roberto Flores (1), Sunil Tholpady (1)
(1) Indiana University, Indianapolis, IN, (2) Indiana University School of Medicine, Indianapolis, IN
Melinda Costa, MD
Generalized protocols in the treatment of Robin Sequence (RS) consider the presence of laryngomalacia as a contraindication to mandibular distraction osteogenesis (MDO). Therefore, little data exist on outcomes in this setting. To avoid tracheostomy, distraction was performed in 11 infants with RS and laryngomalacia; this experience is reported here.
An 8-year (2005-2013) retrospective review of infants with RS and laryngomalacia who underwent MDO at a tertiary care children's hospital was performed. Patients were excluded if they possessed a lower airway anomaly other than laryngomalacia, including tracheomalacia, tracheal web or ring, subglottic stenosis, and vocal cord paralysis. Laryngomalacia was identified on laryngoscopy prior to MDO. Laser supraglottoplasty was performed at the discretion of the otolaryngologist. Variables collected included age at distraction, preoperative and postoperative AHI, syndromic diagnosis or genetic anomalies, cardiac, central nervous system (CNS), and gastrointestinal (GI) abnormalities.
The primary outcomes measured were avoidance or decannulation of tracheostomy and decrease in post-operative AHI.
Eleven infants met inclusion criteria. Mean follow up was 28 months. 18.2% of patients had a syndromic diagnosis, 36.4% cardiac, 9.1% CNS, and 72.7% GI abnormalities. Mean age at time of MDO for patients without prior tracheostomy was 1.7 mo. 60% of patients underwent supraglottoplasty. Mean preoperative AHI was 50.3±32.3 and mean postoperative AHI was 7.7±4.8 (p=0.002). The mean difference between preoperative and postoperative AHI was 42.7±35.2. All patients without a tracheostomy prior to intervention avoided tracheostomy after MDO. One patient had a tracheostomy prior to MDO and was subsequently decannulated. One patient died 1 year after MDO due to complex congenital heart disease.
This study describes a series of patients with RS and laryngomalacia treated successfully with MDO. To our knowledge this is the first report of patients undergoing MDO in the setting of laryngomalacia. These data suggest that infants with laryngomalacia should not be discounted from having MDO to treat upper airway obstruction.