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CE: A.B.; SCS-17-0918; Total nos of Pages: 5;
SCS-17-0918
CLINICAL STUDY
Rigid External Distractor-Aided Advancement After
Simultaneously Performed LeFort-III Osteotomy and
Fronto-Orbital Advancement
Ali-Farid Safi, MD, DMD, Matthias Kreppel, MD, DMD, Martin Kauke, MD,
Andrea Grandoch, MD, DMD, Hans-Joachim Nickenig, DMD, and Joachim Zöller, MD, DMD
Abstract: Due to the complex development of the craniofacial
skull, corrective surgery is a major challenge for patients with
severe craniofaciostenosis. Although fronto-orbital advancement
and simultaneous LeFort-III osteotomy in combination with distraction osteogenesis have been reported as a safe and successful
method to obtain good esthetic and functional results, there is a lack
of studies evaluating this method.
Our retrospective study included 12 patients with syndromic
craniofaciostenosis, who were primarily treated at our department
in accordance with a standardized treatment protocol, consisting of
a simultaneous fronto-orbital advancement with LeFort-III osteotomy in combination with a rigid external distractor (RED-II).
Distraction distance, duration of operation, postoperative complications, perioperative hemoglobin concentration, esthetic outcome,
and the subjective Whitaker Scale were used to evaluate the success
of our surgical method.
The esthetic outcome of all of our patients was assessed as good.
Furthermore, the surgical outcome was assigned I for 11 patients
and II for 1 patient, who suffered from wound healing disturbance at
the left temporal site, which required revision 2 weeks postoperatively. The mean skeletal advancement of the midface was
16.4 mm, ranging from 12 to 20 mm.
Our standardized treatment protocol, consisting of fronto-orbital
advancement in combination with LeFort-III osteotomy and application of a rigid external distractor device (RED-III) for patients
with severe syndromic craniofaciostenosis, goes along with low
infection rates and more predictable and precise esthetic and
functional outcomes than the conventional surgical technique without distraction osteogenesis.
Key Words: Advancement, distraction, fronto-orbital, LeFort,
midfacial
(J Craniofac Surg 2017;00: 00–00)
From the Department for Oral and Craniomaxillofacial Plastic Surgery,
University of Cologne, Cologne, Germany.
Received June 16, 2017.
Accepted for publication August 2, 2017.
Address correspondence and reprint requests to Ali-Farid Safi, MD, DMD,
Department for Oral and Craniomaxillofacial and Plastic Surgery,
University of Cologne, Kerpener Straße 62, 50931 Cologne, Germany;
E-mail: [email protected]
The authors report no conflicts of interest.
Copyright # 2017 by Mutaz B. Habal, MD
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000004061
The Journal of Craniofacial Surgery
P
atients with syndromic craniofaciostenosis frequently suffer
from midfacial hypoplasia and present with esthetic impairments and severe functional problems, for example, intracranial
hypertension, airway obstruction, or exophthalmos leading to corneal ulcerations.1 The surgical treatment of these patients is still a
matter of debate and is mainly based on a fronto-orbito-maxillary
advancement, as proclaimed by Tessier in 1971 or the monobloc
frontofacial advancement, as described by Ortiz-Monasterio in
1978.2,3 However, since the introduction of these methods, the
preeminent complications were high infection rates associated with
contamination from the nasopharynx and the large area of frontal
dead space after advancement.4 Although Whitaker reported that
communication between nasal fossae and intracranial vault might
be avoided by preparation of a mucoperiosteal barrier, Marchac et al
indicated that within their patient cohort, especially for those with a
large advancement, the mucosa ruptured perennially despite consideration of the aforementioned technique.5,6 Therefore, to overcome the severe risks of a 1-step surgical intervention, a 2-stage
procedure regarding the different growth patterns of the neuro- and
viscerocranium was recommended.6,7 This included an early
fronto-orbital advancement and a later LeFort-III osteotomy to
correct the midfacial hypoplasia.6,7 Consequently, recommendation
for the 1-stage approach was generally restricted to severe craniofaciostenosis patients, being associated, for example, with strong
airway obstructions or corneal ulcerations.1 However, since in
1992, McCarthy introduced distraction osteogenesis to craniomaxillofacial surgery, a large number of studies applied this technique
and highlighted a larger and safer skeletal transposition in terms of
fewer recurrences and lower infection rates.8 –13 The authors mainly
explained their results with the limited frontal dead space, the
gradual expansion of the soft tissue, and the ability of re-epithelialization of the mucosa.8,14,15 Compared with the frontofacial monobloc advancement with application of the gradual distraction
method, the fronto-orbital advancement with LeFort-III distraction
using an external distractor offers the ability to reconstruct the
neurocranium and the hypoplastic midface separately.11 In 2004,
Kübler et al reported from a cohort of 6 patients that this method
was associated with good esthetic results and a low complication
rate.11 Similarly, in 2012, Medra et al, who performed the same
method, resulted from their cohort of 9 patients with syndromic
craniofaciostenosis, a significant skeletal advancement, which led
to good esthetic and functional results.16
Within our report, we present our experiences with 12 patients
suffering from severe craniofaciostenosis, who were treated with a
standardized treatment protocol, based on a fronto-orbital advancement with LeFort-III osteotomy and application of an external
distraction device (RED-II; KLS Martin, Tuttlingen, Germany).
MATERIALS AND METHODS
Our investigation followed the guidelines of the Helsinki Declaration. The retrospective study included 12 syndromic
Volume 00, Number 00, Month 2017
1
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
CE: A.B.; SCS-17-0918; Total nos of Pages: 5;
SCS-17-0918
The Journal of Craniofacial Surgery
Safi et al
1
2
3
4
5
6
7
8
9
10
11
12
Syndrome
Morbus
Morbus
Morbus
Morbus
Morbus
Morbus
Morbus
Morbus
Morbus
Morbus
Morbus
Morbus
Apert
Crouzon
Crouzon
Crouzon
Crouzon
Apert
Crouzon
Crouzon
Crouzon
Apert
Crouzon
Crouzon
Age at Operation, y
Sex
Follow-Up
3
8
5
11
8
3
9
3
2
6
5
4
Female
Male
Female
Female
Male
Female
Male
Male
Female
Female
Male
Female
42
65
66
54
65
71
38
59
47
51
18
33
Volume 00, Number 00, Month 2017
segment was repositioned and moved anteriorly. After fixation of
the position with miniplates, the frontal bone cap was adapted to the
shape-altered orbital segment and attached to it with wires or
microplates. The space required for the brain was gained by the
fronto-orbital advancement.
TABLE 1. Patient’s Clinical Characteristics
Patients
LeFort-III Osteotomy
The osteotomy of the viscerocranium was performed by using
the same coronal incision approach. Our osteotomy lines included
the zygomatic arch, sphenozygomatic suture, posterior floor of the
orbit, lacrimomaxillary suture, and the nasal bone. Similar to the
fronto-orbito-nasal advancement by Whitaker et al, we developed a
barrier between nasal fossae and intracranial vault, by leaving 3 to
5 mm of the nasal root untouched.5 Subsequently, the pterygoid
plates were separated by insertion of a chisel below the zygomatic
arch. If necessary, an additional mobilization or osteotomy of
incomplete fractured lines was achieved by a caudal rotation of
the facial skull with Rowe forceps. Afterward, 2 microplates were
placed paranasal and two 0.5-mm stainless steel wires were twisted
and fixed to the maxilla and passed through the skin in the region of
the nasolabial folds to optimize the cosmetic outcome. After
extensive blood stasis, osteosynthesis of the bone segments and
suturing of the coronal incision skin line, the horseshoe-shaped
RED Distractor was applied to the cranial vault. For at least
24 hours, suction drains were placed subcutaneously. All patients
received perioperative antibiotics, analgetic medication, and corticosteroids in accordance to their age and weight.
craniofaciostenosis patients, who were primarily treated at our
department (see Table 1).
All patients were assessed by a multidisciplinary team preoperatively and postoperatively on a weekly basis until removal of the
distraction device. Accessory evaluation included standard preoperative hematologic diagnostics, ophthalmologic examination, and
photo documentation of the patient’s dysmorphic craniofacial
system. Postoperative evaluation included hematologic analysis
and supervision by a pediatric specialist for 1 day on the intensive
care unit as well as for the remaining days of their postoperative
hospitalization. In addition, each patient was reevaluated by our
head physician and surgeon.
For the rest of the first postoperative year, follow-up was
performed every 3 months. After this, patients were seen depending
upon their clinical situation either twice or once a year. Patient’s
outcomes are listed in Table 2.
Distraction Osteogenesis Protocol
After a period of 7 days, the distraction was started with
2 0.5 mm per day with a slight overcorrection of 3 to 5 mm at
the dental occlusion level due to possible relapses, as recommended
by a several authors.1,11,16 One of the main advantages of external
distraction devices is the ability to change the distraction vector
during the treatment period. Therefore, we adapted distraction to the
profile, clinical situation, and the dental occlusion. The decision for
the required amount of distraction was mainly based upon clinical
evaluation of the profile and dental occlusion and less on numerical
values obtained by radiologic examinations.11
During a retention period of 6 weeks, the distractor and the wires
were left in situ without further pull activation. Subsequently, the
wires and the RED distractor were usually removed again under
local anesthesia.
Treatment Protocol
Surgery was performed mainly by a craniomaxillofacial surgery
team with the assistance of a neurosurgeon.
Fronto-Orbital Advancement
Coronal incision served as approach to the craniofacial skeleton
in order to perform a standardized bilateral fronto-orbital advancement.17 After osteotomy and removal of the bone segments, the
fronto-orbital bandeau was shaped. The orbital segment was
slightly bent and adapted to the cranial vault after bone incision
in the central part of the forehead. Hence, the frontal bone and
temporal region could be aligned. The newly formed position and
shape were stabilized by microplates. Subsequently, the orbital
Evaluation of the Outcome
Distraction distance, duration of operation, postoperative complications, and perioperative hemoglobin concentration were used
to evaluate the success of our surgical method. The surgical
TABLE 2. Patient’s Outcome
Patients
Midface Distraction
Distance, mm
1
2
3
16
16
20
4
5
6
7
8
9
10
11
12
2
Complication
Preoperative
Weight, kg
Preoperative HB
Concentration (Range), g/dL
Postoperative HB
Concentration (Range), g/dL
Duration of
Operation, min
Esthetic
Outcome
Whitaker
Score
7.4
7.7
9.7
10
14
11
8
9.5
10.7
200
185
197
Good
Good
Good
I
I
II
20
6
12.3
11.8
212
Good
I
17
14
16
12
16
18
7.1
8.5
6.1
7.2
8
8.7
12.1
11.5
10
12
12.7
11
10.5
10
9
10
10
10
207
157
162
178
182
210
Good
Good
Good
Good
Good
Good
I
I
I
I
I
I
16
16
10.3
6.9
12
11.3
11
9.8
189
200
Good
Good
I
I
Wound healing disturbance
temporal left
#
2017 Mutaz B. Habal, MD
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
CE: A.B.; SCS-17-0918; Total nos of Pages: 5;
SCS-17-0918
The Journal of Craniofacial Surgery
Volume 00, Number 00, Month 2017
Fronto-Orbital Advancement
AQ1
outcome was assessed with the subjective Whitaker Score Scale at
the endpoint of the follow-up (ranging from 18 to 71 months)
(Whitaker Category I: No further treatment desirable; Category II:
Alternative bone work required, such as LeFort I or malar augmentation; Category III: Reposition necessary, such as fronto-orbital
advancement or LeFort III distraction; Category IV: Major craniofacial procedure necessary, duplicating or exceeding the original
operation).18 In accordance to previously performed questionnaires
on the evaluation of the esthetic outcome after craniomaxillofacial
surgical procedures, the surgeon, the parents of the patients, and a
resident in craniomaxillofacial plastic surgery were asked to assess
postoperative photographs in the sagittal, frontal, and axial view
from the 1-year follow-up examination.16 If all were satisfied with
the result, the esthetic result was classified as good and poor if either
the parents, the surgeon, or the resident were unsatisfied.16
RESULTS
Our study included 12 patients with a mean age of 5.77 years (range
from 2 to 11 years). Of our patients, 58% were female and 42% male
(see Table 1). The mean follow-up was 50.75 months with a
standard deviation of 16 months and the median follow-up 52.5
months, ranging from 18 to 71 months.
All patients suffered from airway obstructions leading to snoring
and mouth breathing. Severe proptosis was present in all of our
patients and a main inclusion criterion. The dental occlusion was for
all of our patients assigned as class III. Furthermore, 1 of our
patients suffered from open bite. Previous craniofacial operations
were not performed in any of our patients. Clinical examination
confirmed the typical symptoms of patients with severe craniofacial
stenosis. Hence, within our cohort, brachycephaly, midfacial hypoplasia, and incompetent lips were diagnosed for all patients. Furthermore, neurologic evaluation confirmed intracranial
hypertension within our cohort, due to craniofaciostenosis. Postoperatively, we could observe that airway obstructions manifesting,
for example, as snoring, were diminished within all of our patients
during their follow-up. Ophthalmologic examination confirmed the
success of operation, as corneal erosions and papilledema were not
present anymore in any of our patients. The Angle-Class III
occlusion could be transformed into Angle-Class I due to the
distraction treatment protocol and the ability to change and/or to
rectify the distraction vector.
According to the parents of the patients, the surgeon, and the
resident in craniomaxillofacial surgery, the esthetic outcome of all
of our patients was assessed as good (see Fig. 1A-D). Furthermore,
the surgical outcome was assigned I for 11 patients and II for 1
patient, who suffered from wound-healing disturbance at the left
temporal site, which required revision 2 weeks postoperatively.
Our distraction osteogenesis treatment protocol led to a mean
skeletal advancement of the midface of 16.4 mm, ranging from 12 to
20 mm.
DISCUSSION
Since the introduction of craniofacial surgery in the 1960s by
Tessier, major problems for patients, affected with craniofacial
deformities, were on the one hand the high operative risk based
upon the technical circumstances at that time and on the other hand
the high infection risk, due to the developing communication
between the nasopharynx and the intracranial vault after frontoorbital advancement.19 Therefore, reported complication rates associated with the 1 stage fronto-orbito-maxillary advancement by
Tessier and the monobloc frontofacial advancement by Ortiz-Monasterio led to the recommendation to restrict these 1-step procedures
only to severe patients with craniofaciostenosis, for example, for
patients suffering from strong airway obstructions or proptosis
#
2017 Mutaz B. Habal, MD
FIGURE 1. Photographs of female patient at 6 months (top left), 14 months
(top right), and 5 years of age (bottom left and right).
leading to corneal ulcerations.6 However, with the rise of distraction
osteogenesis, as first applied by Codivilla and popularized by
Ilizarov, substantial efforts could be made for reconstructive surgery handling with bone deformities, describing it as a relatively
safe lengthening method.10,20 For craniomaxillofacial surgery, the
most favorable advantage of distraction osteogenesis is the possibility to minimize the frontal epidural dead space, which fills with
blood and therefore is more susceptible to infection in terms of
communication between the nasopharynx and the intracranial
cavity.21 In 1997, Sugawara et al described their experience on
gradual cranial vault expansion for the treatment of craniofacial
synostosis and reported good results without observing any complications in 3 patients.4 Furthermore, Hirabayashi et al indicated
that fronto-orbital advancement and gradual distraction are associated with a clinically irrelevant deterioration of the dura and a
minimal resorption of the advanced bone, thus minimizing
relapse.22 They highlighted that distraction obviates the need for
bone or skin grafts and therefore is a recommendable corrective
surgical technique.22 However, in accordance with the recommendation of Tessier, both Sugawara et al and Hirabayashi et al
suggested to limit the distraction distance to 10–12 mm in order
to avoid a large dead space.2 Moreover, Hirabayashi et al indicated
that usage of an external distraction device goes along with scars, a
second operation to remove the device, a relatively long treatment
and a possible infection at the site of the device.22 In 2007, Pelo et al
compared within a literature survey, internal and external craniofacial distractor devices and concluded that although both methods
are effective, significant differences exist concerning the surgical
aspects, and the practical differences experienced by the patients.23
They indicated that for severe patients external devices go along
with a better control of the vectors and assure more predictable and
3
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
CE: A.B.; SCS-17-0918; Total nos of Pages: 5;
SCS-17-0918
Safi et al
The Journal of Craniofacial Surgery
effective final results.23 Hence, for our patients, we used a rigid
external distraction device (RED-II), first described by Polley et al
in 1998, in order to modify the vector in all 3 dimensions.24 By
placing the wires near to the piriform aperture, the distraction forces
can be applied in the central part of the facial skeleton, thus enabling
better cosmetic results than anchoring the wire on the teeth or the
lateral part of the zygoma.11 Compared with the monobloc frontofacial advancement, proclaimed by Ortiz-Monasterio in 1978, the
main advantage of the fronto-orbital advancement and LeFort-III
osteotomy is the ability to correct both areas separately and thus
have more options to correct the midfacial hypoplasia and occlusion.3,6,17,25 However, advocates of the combination of frontofacial
monobloc advancement with gradual distraction argue that due to
the larger monobloc segment, the distraction force is much greater
than it is for the LeFort-III segment.21 Kamoshima et al, for
example, stated that within their cohort of 3 patients, who underwent a frontofacial monobloc advancement using the gradual
distraction method the mean lengths ranged from 19 to 25 mm.21
Witherow et al indicated for their study group of 21 patients that the
midface could be distracted at a mean of 16.4 mm with a range from
12 to 22 mm.1 However, we also achieved advancements of the
midface of up to 20 mm with a mean distraction distance of
16.40 mm. Therefore, the esthetic and surgical outcomes were good
and did not necessitate larger skeletal transpositions, as for example
described by Kamoshima et al.21 Our distraction distances were
similar to those reported by Kübler et al with a mean rate of
15.8 mm and a maximum distance of 18 mm.11 In 2012, Medra
et al indicated that the clinical distraction distance within their patient
cohort ranged from 15 to 20 mm with a mean transposition of 18 mm.
Both Kübler et al and Medra et al reported low complication rates and
good esthetic outcomes using a standardized treatment protocol
similar to ours. In 1 of our patients, we could observe wound-healing
disturbances, which necessitated reoperation 2 weeks postoperatively. For the rest of our patients, no complications particularly
infections were diagnosed. This finding is in accordance with the
majority of published literature, who reported low complication rates
using external distraction devices in combination with advancement
of the fronto-orbital region and the midface.1,23 Furthermore, as
Whitaker et al reported that by means of a sufficiently deep or basal
osteotomy in the area of the nose/frontobase the risk for ascending
infections might be reduced, we assume that application of this
technique played also an important role for the low complication
rate within our study.5 A further important factor to prevent infection
is that distraction should start from the 7th postoperative day so that
the soft tissue in the area of the frontal skull is given enough time to
consolidate and thereby adhere to the adjacent bone.11,26 Thus,
although several disadvantages of external distraction devices are
reported within literature, we could not observe infections at the
exposed site of the device and obtained good results with the method
of fronto-orbital advancement with LeFort-III osteotomy in combination with a rigid external distractor (RED-II).
Another potential factor for infection is the usage of titanium
plates. Fixation of the newly shaped neuro- and viscerocranium
must be enabled by a rigid system, which is reliable in terms of
stability, ductility, and biocompatibility.27,28 Since, in the late
1980s, titanium plates were introduced into craniomaxillofacial
surgery, they were frequently used for fixation and led to excellent
results.27,28 However, some authors noticed a risk of growth
restriction, transcranial migration and highlighted a higher surgical
risk, as removal of the titanium plates in a second surgical procedure
is necessary.27,28 Therefore, with the rise of biodegradable polymeric materials in the mid-1990s, a second intervention could be
avoided and therefore are nowadays favored.28,29 However, some
reports on resorbable plates indicated a potential risk of inflammatory reaction, incomplete resorption, granuloma formation,
4
Volume 00, Number 00, Month 2017
osteolysis at the location of the plates, sterile fistula formation,
or sterile abscesses.29–31 At our department, we routinely use
titanium plates in all of our patients and remove these in a second
operation regularly after approximately 5 to 6 months. In our study,
we could not observe any complications related to the titanium
plates, for example, growth-induced intracranial migration or infection at the plate sites, as our protocol is specifically intended to
reduce the risk of such sequelae.
CONCLUSION
Our standardized treatment protocol, consisting of fronto-orbital
advancement in combination with LeFort-III osteotomy and application of a rigid external distractor device (RED-III) for patients with
severe syndromic craniofaciostenosis, goes along with low infection
rates and more predictable and precise esthetic and functional outcomes
than the conventional surgical technique without distraction osteogenesis. However, further studies conducted on larger study groups and
long-term follow-up results are needed to evaluate our findings.
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#
2017 Mutaz B. Habal, MD
Copyright © 2017 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
CE: A.B.; SCS-17-0918; Total nos of Pages: 5;
SCS-17-0918
The Journal of Craniofacial Surgery
Volume 00, Number 00, Month 2017
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Fronto-Orbital Advancement
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5
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