71 / 280
results were essentially unchanged, suggesting minimal im-
pact of this possible bias. Regardless, the issue of confound-
ing by indication is an important one, which is why the out-
comes of surgical successwere addressed in separate analyses
in this study. Within the context of this broad retrospective
study, we did not attempt to directly compare the 2 primary
interventions of tracheotomy andMDO except with respect to
overall rates of complications. However, it is clear from our
analysis that patients who were syndromic or had additional
airway disease abnormalities were more likely to have an ini-
tial tracheotomy to addressmultilevel disease.Mandibular dis-
traction osteogenesis should be considered only as an initial
procedure in a setting inwhich there is no other indication for
tracheotomy, such as multilevel airway obstruction, ventila-
tor dependence, or persistent dysphagia and aspiration. Be-
cause patients with CFM-Goldenhar syndrome were found to
have independently lower odds of successwithMDOthanother
syndromic conditions,wewouldnot recommendprimaryMDO
in these patientswhohave clinically significant respiratorydis-
tress due tomicrognathia. A proposed treatment algorithm is
presented in
Figure 3
.
Conclusions
Our study evaluated a large cohort of children that had under-
gone MDO. In our study we demonstrated a high rate of sur-
gical success for MDO with a low rate of complications, par-
ticularly among patients treated with distraction as an initial
procedurewithout anexisting tracheotomy. However, it is clear
that patients treatedwith tracheotomy initially aremore likely
to be syndromic and complex, requiring greater numbers of
distractions and airway procedures. In addition, patients with
CFM–Goldenhar syndromehave a decreasedprobabilityof sur-
gical success comparedwith patients with other types of syn-
dromes or nonsyndromic Pierre Robin sequence.
ARTICLE INFORMATION
Submitted for Publication:
September 6, 2013;
final revision received December 14, 2013; accepted
January 6, 2014.
Published Online:
February 27, 2014.
doi:10.1001/jamaoto.2014.16.
Author Contributions:
Dr Lam had full access to all
of the data in the study and takes responsibility for
the integrity of the data and the accuracy of the
data analysis.
Study concept and design:
Lam, Shikary,
Uribe-Rivera, de Alarcon, Gordon.
Acquisition of data:
Lam, Shikary, Uribe-Rivera.
Analysis and interpretation of data:
Lam, Tabangin,
Shikary, Meinzen-Derr, Billmire.
Drafting of the manuscript:
Lam, Meinzen-Derr.
Critical revision of the manuscript for important
intellectual content:
Lam, Tabangin, Shikary,
Uribe-Rivera, de Alarcon, Billmire, Gordon.
Statistical analysis:
Lam, Tabangin, Shikary,
Meinzen-Derr.
Administrative, technical, and material support:
Uribe-Rivera, Billmire.
Study supervision:
Lam, de Alarcon, Billmire,
Gordon.
Conflict of Interest Disclosures:
None reported.
Previous Presentation:
This study was presented
at the American Society of Pediatric Otolaryngology
Spring Meeting; April 19-22, 2012; San Diego,
California.
REFERENCES
1
. Robin P. A fall of the base of the tongue
considered as a new cause of nasopharyngeal
respiratory impairment: Pierre Robin sequence,
a translation: 1923.
Plast Reconstr Surg
.
1994;93(6):1301-1303.
2
. Meyer AC, Lidsky ME, Sampson DE, Lander TA,
Liu M, Sidman JD. Airway interventions in children
with Pierre Robin sequence.
Otolaryngol Head Neck
Surg
. 2008;138(6):782-787.
3
. Sedaghat AR, Anderson IC, McGinley BM,
Rossberg MI, Redett RJ, Ishman SL.
Characterization of obstructive sleep apnea before
and after tongue-lip adhesion in children with
micrognathia.
Cleft Palate Craniofac J
.
2012;49(1):21-26.
4
. Denny AD, Amm CA, Schaefer RB. Outcomes of
tongue-lip adhesion for neonatal respiratory
distress caused by Pierre Robin sequence.
J Craniofac Surg
. 2004;15(5):819-823.
5
. Kremer B, Botos-Kremer AI, Eckel HE,
Schlöndorff G. Indications, complications, and
surgical techniques for pediatric tracheostomies:
an update.
J Pediatr Surg
. 2002;37(11):1556-1562.
6
. Perkins JA, Sie KC, Milczuk H, Richardson MA.
Airway management in children with craniofacial
anomalies.
Cleft Palate Craniofac J
.
1997;34(2):135-140.
7
. Rachmiel A, Srouji S, Emodi O, Aizenbud D.
Distraction osteogenesis for tracheostomy
dependent children with severe micrognathia.
J Craniofac Surg
. 2012;23(2):459-463.
8
. Mudd PA, Perkins JN, Harwood JE, Valdez S,
Allen GC. Early intervention: distraction
osteogenesis of the mandible for severe airway
obstruction.
Otolaryngol Head Neck Surg
.
2012;146(3):467-472.
9
. Hammoudeh J, Bindingnavele VK, Davis B, et al.
Neonatal and infant mandibular distraction as an
alternative to tracheostomy in severe obstructive
sleep apnea.
Cleft Palate Craniofac J
.
2012;49(1):32-38.
10
. Tibesar RJ, Scott AR, McNamara C, Sampson D,
Lander TA, Sidman JD. Distraction osteogenesis of
the mandible for airway obstruction in children:
long-term results.
Otolaryngol Head Neck Surg
.
2010;143(1):90-96.
11
. Senders CW, Kolstad CK, Tollefson TT, Sykes JM.
Mandibular distraction osteogenesis used to treat
upper airway obstruction.
Arch Facial Plast Surg
.
2010;12(1):11-15.
12
. Miloro M. Mandibular distraction osteogenesis
for pediatric airway management.
J Oral Maxillofac
Surg
. 2010;68(7):1512-1523.
13
. Tibesar RJ, Sidman JD. Mandibular distraction
osteogenesis in the pediatric patient.
Curr Opin
Otolaryngol Head Neck Surg
. 2008;16(6):548-554.
14
. Zim S. Treatment of upper airway obstruction in
infants with micrognathia using mandibular
distraction osteogenesis.
Facial Plast Surg
.
2007;23(2):107-112.
15
. Mandell DL, Yellon RF, Bradley JP, Izadi K,
Gordon CB. Mandibular distraction for micrognathia
and severe upper airway obstruction.
Arch
Otolaryngol Head Neck Surg
. 2004;130(3):
344-348.
16
. McCarthy JG, Schreiber J, Karp N, Thorne CH,
Grayson BH. Lengthening the human mandible by
gradual distraction.
Plast Reconstr Surg
.
1992;89(1):1-8.
17
. Denny AD, Talisman R, Hanson PR, Recinos RF.
Mandibular distraction osteogenesis in very young
patients to correct airway obstruction.
Plast
Reconstr Surg
. 2001;108(2):302-311.
18
. Scott AR, Tibesar RJ, Lander TA, Sampson DE,
Sidman JD. Mandibular distraction osteogenesis in
infants younger than 3 months.
Arch Facial Plast
Surg
. 2011;13(3):173-179.
19
. Ow AT, Cheung LK. Meta-analysis of mandibular
distraction osteogenesis: clinical applications and
functional outcomes.
Plast Reconstr Surg
.
2008;121(3):54e-69e.
20
. Kolstad CK, Senders CW, Rubinstein BK,
Tollefson TT. Mandibular distraction osteogenesis:
at what age to proceed.
Int J Pediatr
Otorhinolaryngol
. 2011;75(11):1380-1384.
21
. Evans AK, Rahbar R, Rogers GF, Mulliken JB,
Volk MS. Robin sequence: a retrospective review of
115 patients.
Int J Pediatr Otorhinolaryngol
.
2006;70(6):973-980.
22
. Evans KN, Sie KC, Hopper RA, Glass RP, Hing
AV, CunninghamML. Robin sequence: from
diagnosis to development of an effective
management plan.
Pediatrics
. 2011;127(5):
936-948.
23
. Rogers GF, Lim AA, Mulliken JB, Padwa BL.
Effect of a syndromic diagnosis on mandibular size
and sagittal position in Robin sequence.
J Oral
Maxillofac Surg
. 2009;67(11):2323-2331.
Mandibular Distraction Osteogenesis
Original Investigation
Research
jamaotolaryngology.comJAMA Otolaryngology–Head & Neck Surgery
April 2014
Volume
140, Number 4
49