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around 1:8,500,3,4 there are approximately 500 new cases of
PRS in the US annually, 30% of which may have airway
obstruction severe enough to warrant operative intervention.11
Our data suggest a $300,000 cost savings for each patient
treated with mandibular distraction rather than tracheostomy,
over 3 years. Hypothetically, performing MD on all patients
with severe PRS would generate health care savings of
$450,000,000 over a 10-year period, assuming those patients
would otherwise receive a tracheostomy. This estimate does
not include long-term and indirect costs, which include costs
associated with scars, radiation exposure, and quality of life.
Cost savings is one important consideration for selection
of treatment of PRS and is the focus of this study. Of equal
or greater importance though are the long-term airway,
speech, nutrition, and developmental outcomes of patients
treated with mandibular distraction compared to tracheost-
omy. However, as described in a recent systematic review,
outcomes of neonates with PRS are poorly understood due
to wide variability of study inclusion criteria, lack of stan-
dardized indications for interventions, and a general paucity
of data.5 Based on the present study and our clinical experi-
ence,10 we support the use of mandibular distraction for
treatment of severe PRS but acknowledge that further study
is necessary to standardize diagnostic and treatment criteria
and to identify appropriate outcome measures.
Acknowledgments
We thank Dr David Billmire, Dr J. Paul Willging, Dr Evan Probst,
and Patricia Bender for assistance with study design, data collec-
tion, and analysis.
Author Contributions
Christopher M. Runyan, data collection, data analysis, majority of
the writing; Armando Uribe-Rivera, data collection, data analysis,
writing; Audrey Karlea, data collection, data analysis, early manu-
script writing; Jareen Meinzen-Derr, statistical and data analyses;
Dawn Rothchild, data collection, data analysis, early manuscript revi-
sions; Howard Saal, study conception and design, data collection, data
analysis, early manuscript revisions; Robert J. Hopkin, study concep-
tion and design, data collection, data analysis, Institutional Review
Board protocol, early manuscript draft and revisions; Christopher B.
Gordon, study conception and design, data collection, data analysis.
Disclosures
Competing interests: None.
Sponsorships: None.
Funding source: Internal, we would like to acknowledge the
Cincinnati Children’s Hospital Medical Center Divisions of
Genetics and Plastic Surgery for funding this project.
Supplemental Material
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