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Original Research—Health Policy and Economics
Cost Analysis of Mandibular Distraction
versus Tracheostomy in Neonates with
Pierre Robin Sequence
Otolaryngology–
Head and Neck Surgery
2014, Vol. 151(5) 811–818
American Academy of
Otolaryngology—Head and Neck
Surgery Foundation 2014
Reprints and permission:
sagepub.com/journalsPermissions.navDOI: 10.1177/0194599814542759
http://otojournal.orgChristopher M. Runyan, MD, PhD
1
*
, Armando Uribe-Rivera
1
*
,
Audrey Karlea
2
, Jareen Meinzen-Derr, PhD
3
, Dawn Rothchild
1
,
Howard Saal, MD
4
, Robert J. Hopkin, MD
4§
, and
Christopher B. Gordon, MD
1§
Sponsorships or competing interests that may be relevant to content are dis-
closed at the end of this article.
Abstract
Objective
. To evaluate costs associated with surgical treat-
ment for neonates with Pierre Robin sequence (PRS).
Study Design
. Retrospective cohort study.
Setting
. Cincinnati Children’s Hospital Medical Center.
Subjects and Methods
. With Institutional Review Board
approval, we retrospectively studied neonates with PRS
treated from 2001 to 2009 with either tracheostomy (Trach),
mandibular distraction (MD), or Trach with subsequent MD
(Trach
1
MD). Actual charges over a 3-year period associated
with operative costs, hospital stay, imaging and sleep studies,
clinic visits, and related emergency room visits were col-
lected. Home tracheostomy care charges were estimated
individually for each patient. Charges were compared using
regression and appropriate statistical analyses.
Results
. Forty-seven neonates were included in the study
(MD, n = 26; Trach, n = 12; Trach
1
MD, n = 9). Trach group
patients had 2.6-fold higher charges than the MD group
despite no difference in length of hospital stay. This difference
increased to 7.3-fold when including home trach care-related
costs. Trach
1
MD group patients had longer hospital lengths
of stay and higher operation room (OR) fees, but no
increased total charges compared with the Trach only group.
Conclusions
. For patients with severe PRS, mandibular distrac-
tion provides significant cost savings over tracheostomy
($300,000 per patient over 3 years). Increased costs with tra-
cheostomy come from greater hospital-related charges, more
frequent airway procedures, a higher incidence of gastrostomy
tube feeds, and home trach care costs. A careful examination
of long-term outcomes will be critical as mandibular distraction
continues to gain acceptance for treatment of PRS.
Keywords
Pierre Robin sequence, tracheostomy, mandibular distrac-
tion osteogenesis
Received January 29, 2014; revised May 13, 2014; accepted June 18,
2014.
Introduction
Defined by the triad of micrognathia, glossoptosis, and
airway obstruction,
1
Pierre Robin sequence (PRS) is a
morbid and potentially lethal condition among neonates. In
PRS, mandibular hypoplasia leads to abnormal cephalad and
posterior positioning of the tongue, which frequently results
in failure of fusion of the secondary palate, and a U-shaped
cleft palate.
2
PRS occurs in 1:8500 live births and may
occur in isolate or with a genetic syndrome, most commonly
Stickler and Velocardiofacial syndromes, and hemifacial
microsomia.
3,4
Clinically, PRS is marked by oxygen desa-
turations, apnea, gastroesophageal reflux, feeding difficul-
ties, and failure to thrive. Mortality rates associated with
PRS range from 0% to 21% (median 4.5%).
5
Mild cases
may be managed conservatively, using prone positioning,
nasopharyngeal or laryngeal mask airways, or palatal
obturators. Moderate to severe cases require surgical inter-
vention to relieve or bypass the airway obstruction.
Surgery for PRS consists of tongue-lip adhesion (TLA),
tracheostomy, or mandibular distraction. Given the
1
Division of Plastic Surgery, Cincinnati Children’s Hospital Medical Center,
Cincinnati, Ohio, USA
2
Kaiser Permanente Clinical Genetics Department, Oakland, California,
USA
3
Division of Biostatistics and Epidemiology, Cincinnati Children’s Hospital
Medical Center, Cincinnati, Ohio, USA
4
Division of Human Genetics, Cincinnati Children’s Hospital Medical
Center, Cincinnati, Ohio, USA
*
These authors contributed equally to this article.
§
These authors contributed equally to this project.
Presented at the 12
th
International Congress on Cleft Lip/Palate and
Related Craniofacial Anomalies; May 5-10, 2013; Orlando, Florida.
Corresponding Author:
Christopher M. Runyan, MD, PhD, Division of Plastic Surgery, Cincinnati
Children’s Hospital Medical Center, 200 Albert Sabin Way, Mail Location
0461, Cincinnati, OH 45219, USA.
Email:
chris.runyan@cchmc.orgReprinted by permission of Otolaryngol Head Neck Surg. 2014; 151(5):811-818.
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