Predicting Failure of Mandibular Distraction Osteogenesis for Infants with Robin Sequence: A Bi-Institutional Study
Melinda A. Costa, MD1, Sunil S. Tholpady, MD, PhD1, Mark M. Urata, DDS, MD2, Jeffrey Hammoudeh, DDS, MD2, Rachel E. Sargent, BA3, Ellynore Florendo, MS3, Luke Sanborn, BA2, Roberto L. Flores, MD4.
1Indiana University, Indianapolis, IN, USA, 2Children's Hospital Los Angeles, Los Angeles, CA, USA, 3Children's Hospital, Los Angeles, Los Angeles, CA, USA, 4New York University, New York, NY, USA.
We combined two institutional experiences to identify variables associated with failure of mandibular distraction osteogenesis (MDO) in infants with Robin sequence (RS) and analyze the GILLS score’s ability to predict MDO outcome. This represents the largest infant distraction experience to date.
A retrospective review (2004 – 2013) of infants <6 months of age with RS undergoing MDO at two tertiary care children’s hospitals. Variables included cardiac, central nervous system (CNS), genetic/syndromic anomalies, laryngomalcia, isolated disease, Nissen fundoplication, intact palate, gastroesophageal reflux disease (GERD), preoperative intubation, late operation (>2 weeks of age), and low birth weight (<2500 g). The GILLS score was calculated. Postoperative tracheostomy was considered treatment failure.
91 consecutive infants were identified. Mean age at distractor placement was 36 days. The failure rate was 6.6% (6 patients). Variables associated with failure were: Intact palate (p <0.0042), Nissen (p <0.0007) and CNS anomaly (p <0.0003). 3 deaths occurred; none related to airway obstruction. The “INC” score was derived based on the three variables associated with MDO failure. For infants with < 3 risk factors, INC predicted success with 99% sensitivity, 98% positive predictive value (PPV), 67% specificity, and 80% negative predictive value (NPV). GILLS scoring resulted in 79% sensitivity, 96% PPV, 50% specificity, and 14% NPV.
In infants with RS, intact palate, need for Nissen fundoplication and CNS anomaly are associated with MDO failure. The INC score better predicts MDO failure compared to the GILLS classification. Prospective analysis should be considered to validate the INC score.
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