66 / 280
sion in subsequentmultivariable regressionmodels. Anα = .05
was considered for statistical significance in all final models.
SAS statistical software (version 9.3; SAS Institute) was used
to conduct all analyses.
Results
There were 132 patients who underwent MDO during the
study period. Of these, 8 patients were lost to follow-up, and
1 died shortly after distraction owing to congenital heart dis-
ease. These patients were excluded from subsequent analy-
sis, leaving 123 patients in the cohort. Patient characteristics
for the entire cohort and stratified by initial treatment group
are described in
Table 1
. A slight majority of patients were
male, and 56.0% were diagnosed as having an associated
syndrome. Treacher-Collins (9.8%) and CFM-Goldenhar
(8.9%) syndromes were the most commonly encountered
syndromes. Median age at time of distraction was 21 months
(range, 6 days–24 years). Sixty-two patients (50.4%) under-
went an initial tracheotomy prior to MDO while 61 (49.6%)
underwent MDO first. Median follow-up time was approxi-
mately 5 years (range, 30 days–16.2 years). The median dis-
traction amount was 22 mm (range, 7-52 mm). One hundred
seven patients (87.0%) underwent a single distraction, while
the remainder of the cohort required repeated distractions.
Most these patients underwent repeated distractions at least
6 months after the initial distraction. Seven patients (5.7%)
required repeated distractions within 30 days of the initial
procedure. Of these, 4 could be attributed to either hardware
malfunction or premature consolidation. Sixty-seven
patients (55.0%) did not require any further airway proce-
dures after distraction, although a substantial minority of
patients required at least 1 other procedure.
Patients who underwent tracheotomy first were signifi-
cantly more likely to have an associated syndromic diagnosis
compared with those who underwent MDO first (66.0%
vs 43.0%, respectively). Patients treated with MDO first
were also significantly younger at the time of distraction
than those treated with tracheotomy first (median age,
5 months vs 30 months) and required fewer subsequent air-
way procedures.
Table 1. Characteristics and Outcomes of 123 Patients in Cohort
Characteristic
No. (%)
Tracheotomy First
(n = 62 [50.4%])
MDO First
(n = 61 [49.6%])
P
Value
Male
69 (56.1)
Isolated Pierre Robin sequence
54 (43.9)
21 (33.9)
35 (57.4)
Syndromic
69 (56.1)
41 (66.3)
26 (42.6)
Treacher-Collins syndrome
12 (9.8)
8 (12.9)
4 (6.6)
.009
CFM–Goldenhar syndrome
11 (8.9)
7 (11.3)
4 (6.6)
Stickler syndrome
4 (3.3)
3 (4.8)
1 (1.6)
Other
a
42 (3.1)
23 (3.7)
17 (2.8)
Age at distraction, mo
Mean (SD)
40 (52)
46 (47)
34 (57)
.20
Median (IQR)
21 (2.2-48.2)
30 (15.2-52.8)
5.1 (0.6-42.1)
<.001
Range
6 d–20 y
25 d–20 y
6 d–19 y
Follow-up time, mean (SD), y
3.2 (3.2)
3.8 (3.8)
2.5 (2.3)
.03
Amount distracted, mean (SD), mm 22.3 (7.9)
23.1 (7.9)
21.6 (7.7)
.28
Distractions, No.
1
107 (87.0)
55 (88.7)
52 (85.2)
.52
c
2
14 (11.4)
7 (11.3)
7 (11.5)
≥3
2 (1.6)
0
2 (3.3)
Subsequent airway procedures, No.
b
0
67 (55.4)
24 (38.7)
43 (72.9)
.002
1
27 (22.3)
18 (29.0)
9 (15.2)
2
10 (8.3)
7 (11.3)
3 (5.1)
≥3
17 (14.0)
13 (21.0)
4 (6.8)
Outcomes by treatment group
Surgical success
93 (75.6)
42 (67.7)
51 (83.6)
Avoidance of tracheotomy
NA
NA
51 (83.6)
<.001
Decannulation
NA
42 (67.7)
5 (8.2)
Complications
33 (26.8)
24 (38.7)
9 (14.8)
.03
Premature consolidation
14 (11.4)
12 (19.4)
2 (3.3)
.005
Open bite deformity
9 (7.3)
5 (8.1)
4 (6.6)
>.99
c
Temporomandibular joint ankylosis
5 (4.1)
5 (8.1)
0
.06
c
Other
d
9 (7.3)
6 (9.7)
3 (5.0)
.49
c
Abbreviations: CFM, craniofacial
microsomia; IQR, interquartile range;
NA, not applicable.
a
Includes arthrogryposis; Pfeiffer,
Nager, Klippel-Feil, amniotic band,
orofaciodigital, Cornelia de Lange,
Loeys-Dietz, Dandy-Walker,
cri-du-chat, Crouzon, Moebius, and
Katel-Manske syndromes;
achondroplasia; and chromosomal
abnormalities.
b
Includes laryngotracheoplasty,
endoscopic airway procedures, base
of tongue procedures, choanal
atresia repair.
c
Fisher exact test.
d
Need for repeated distraction within
30 days of initial distraction,
transient facial nerve injury,
emergent reintubation, prolonged
intubation, and internal carotid
artery dissection.
Research
Original Investigation
Mandibular Distraction Osteogenesis
JAMA Otolaryngology–Head & Neck Surgery
April 2014 Volume 140, Number 4
jamaotolaryngology.com44