ny-

CME

Thirty Years Later: What Has Craniofacial

Distraction Osteogenesis Surgery Replaced?
Richard A. Hopper, M.D.,
M.S.
Russell E. Ettinger, M.D.
Chad A. Purnell, M.D.
M. Stephen Dover, B.D.S.,
M.B.Ch.B.
Alberto Rocha Pereira, M.D.
Gökhan Tunçbilek, M.D.,
Downloaded from http://journals.lww.com/plasreconsurg by BhDMf5ePHKbH4TTImqenVKP86OIkNq69sax+N08DK7uBi3fMCDVLyfGU4E9OoUvBiEiNOOoSv5E= on 05/27/2020
Learning Objectives: After studying this article and viewing the video, the par-
ticipant should be able to: 1. Compare the relative stability and neurosensory
changes following mandible distraction osteogenesis with those after tradi-
tional advancement and fixation. 2. Describe the condylar changes that can
occur after mandible distraction osteogenesis and list three ways to mitigate
these changes. 3. Propose clinical situations where segmental or rotational
movements of the midface may allow improved outcomes compared to en bloc
linear distraction advancement. 4. Summarize the advantages and risks associ-

Ph.D. ated with anterior and posterior cranial distraction osteogenesis compared to
Seattle, Wash.; Birmingham, United traditional one-stage expansion.
Kingdom; Lisbon, Portugal; Summary: Over the past 30 years, distraction forces have been applied to the
and Ankara, Turkey spectrum of craniofacial osteotomies. It is now time to assess critically the cur-
rent understanding of distraction in craniofacial surgery, identifying both tra-
ditional procedures it has replaced and those it has not. This article provides
a review of comparative studies and expert opinion on the current state of cra-
niofacial distraction compared with traditional operations. Through this criti-
cal evaluation, the reader will be able to identify when distraction techniques
are appropriate, when traditional techniques are more favorable, and what the
future of distraction osteogenesis is. (Plast. Reconstr. Surg. 145: 1073e, 2020.)

T
he animal work of Snyder et al. introduced
the possibility of distraction osteogenesis of
the mandible in 1972.1 Joseph McCarthy’s
basic science and clinical studies made it a reality
in the early 1990s.2–4 Over the past 30 years, dis-
traction forces have been applied to the spectrum
of craniofacial osteotomies, but its role continues
to be debated and challenged.5,6 Initially heralded
as a replacement for traditional orthognathic,
grafting, and fixation procedures,7 it is now time
to critically assess the role of distraction in cranio-
facial surgery, identifying both traditional proce-
dures it has replaced and those it has not.
We wrote this continuing education article
not as a general overview of what craniofacial dis-
traction can accomplish. Instead, it is a review of
comparative studies and expert opinion on the
current state of craniofacial distraction compared
with traditional surgery. Through this critical eval-
uation, the reader will be able to identify when
distraction techniques are appropriate, when tra-
ditional techniques are more favorable, and what
the future of distraction osteogenesis is.
MANDIBLE
Mandible Distraction (Mandibular Distraction
Osteogenesis) Surgery to Achieve Final Occlusal
Position
Pioneers in distraction viewed distraction
osteogenesis as a potential replacement for the
traditional sagittal split osteotomy and fixation.7
Mandibular distraction osteogenesis has been

From the Division of Plastic Surgery, University of Wash-
ington; the Craniofacial Center, Seattle Children’s Hospital;
the Craniofacial Unit, Birmingham Children’s Hospital; the
Facial Reconstructive Unit, Hospital da Luz; and the De-
partment of Plastic, Reconstructive and Aesthetic Surgery,
Hacettepe University Faculty of Medicine.
Received for publication March 10, 2019; accepted Septem-
ber 9, 2019.
Copyright © 2020 by the American Society of Plastic Surgeons
DOI: 10.1097/PRS.0000000000006821
Disclosure: Dr. Hopper is an inventor on a patented
nasal molding device licensed for distribution to KLS
Martin LLP. No other authors have financial interests
to disclose. No funding was received for this article.
Related digital media are available in the full-text
version of the article on www.PRSJournal.com.

www.PRSJournal.com 1073e
Copyright © 2020 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.

associated with a 36 percent increase in overall
cost compared with single-stage surgery, primarily
because of the need for a second operation and
device cost.8 It is therefore important to delineate
advantages that would justify the additional cost
of mandibular distraction osteogenesis as a substi-
tute for sagittal split osteotomy.
With a recognized risk of iatrogenic inferior
alveolar nerve injury during sagittal split osteot-
omy,9 there has been a resurgent interest in using
mandibular distraction osteogenesis as the final
occlusal surgery for class II patients. The theory
is that gradual lengthening across the osteotomy
would be less traumatic than an acute movement.
Early systematic reviews reported a lower rate of
neurosensory changes following mandibular dis-
traction osteogenesis compared with sagittal split
osteotomy,10,11 but included only retrospective
studies. There have been no objective neurosen-
sory differences detected in more recent pro-
spective randomized trials between sagittal split
osteotomy and mandibular distraction osteogen-
esis12–14 (Reference 12 Level of Evidence: Thera-
peutic, II).
Studies comparing occlusal stability following
small (<6 mm) advancements in mature patients
have not shown a statistical difference between the
two procedures.10,11 In advancements greater than
10 mm, there is a significant difference, with stabil-
ity demonstrated following mandibular distraction
osteogenesis15 but not following sagittal split oste-
otomy.16,17 Mandibular distraction osteogenesis is
accepted as the only option for extremely large
movements (e.g., ≥20 mm). The intrinsic instabil-
ity of a high mandibular plane angle correction
(counterclockwise rotation), however, remains a
challenge for both techniques. Van Strijen et al.15
found relapse in 57 percent of patients with a
rotated high mandibular plane angle treated with
mandibular distraction osteogenesis compared
with 8 percent in the normal angle group; these
rates were comparable to the 30 percent and 11
percent rates, respectively, observed after sagittal
split osteotomy.14
The role of mandibular distraction osteogen-
esis in immature craniofacial microsomia patients
with mild to moderate mandible deformities
is unclear as a means to achieve final occlusal
position. In 2008, Nagy et al. concluded, “there
is a lack of statistical evidence to support the
use of early distraction osteogenesis for correct-
ing hemifacial microsomia as a single treatment
modality.”18 Similar findings were reported in a
systematic review in 2013 from a lack of random-
ized studies or outcomes at maturity.19 Since that
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Plastic and Reconstructive Surgery • June 2020
time, a long-term outcome analysis of mandibular
distraction osteogenesis in 33 microsomia patients
demonstrated that 90 percent had recurrence of
asymmetry after 5 to 15 years of follow-up that
required definitive orthognathic surgery.20 Zhang
et al. reported a 50 percent need for orthognathic
surgery at maturity between microsomia patients
with Pruzansky type IIB/III mandible deformities,
regardless of whether they underwent earlier dis-
traction21 (Reference 21 Level of Evidence: Ther-
apeutic, III). Weichman et al. subjectively assessed
19 mature patients with mild microsomia (type I/
IIA) who had undergone mandibular distraction
osteogenesis during growth and reported that 12
patients were considered aesthetically satisfactory
and seven unsatisfactory.22 They concluded that
younger age at distraction and overcorrection of
the deformity after surgery were predictors of sat-
isfactory results, but that 41 percent of the satisfac-
tory group and 71 percent of the unsatisfactory
group still required secondary orthognathic or
other major operations. Meazzini et al. followed
14 type I/II microsomia patients to maturity after
early mandibular distraction osteogenesis (aver-
age age, 6 years) and reported a 100 percent
relapse rate.23 In comparison, Lu followed seven
consecutive Pruzansky type II microsomia patients
(aged 12 and 16 years) to maturity following simul-
taneous maxillary-mandibular distraction and
reported that cant correction was stable, along
with other measures of asymmetry.24 From this,
they advocated adolescent double-jaw surgery as a
replacement for orthognathic surgery at maturity.
The literature to date therefore supports man-
dibular distraction osteogenesis as a means to
achieve larger advancements than are possible by
sagittal split osteotomy, but not as a replacement
for more traditional procedures in achieving stable
occlusion at maturity. It remains an unanswered
question whether the bone formed by earlier man-
dibular distraction osteogenesis makes final occlu-
sal surgery easier or more challenging. Recent
randomized trials have not found an improvement
in neurosensory outcomes to justify the increased
cost. In craniofacial microsomia patients, studies
at maturity have found that early mandibular dis-
traction osteogenesis did not prevent the need for
final occlusal surgery,25,26 but double-jaw distrac-
tion in later adolescence may hold promise.
Condylar Changes following Mandibular
Distraction Osteogenesis
Mandibular distraction osteogenesis creates a
bidirectional force across an osteotomy. Although
the desired effect is for the tooth-bearing segment
 Volume 145, Number 6 • Craniofacial Distraction Osteogenesis
Fig. 1. Condylar resorption in a patient with craniofacial microsomia who underwent bilateral mandible distraction with external
devices at age 3. (Left) Preoperative image at age 3 years, with noted Pruzansky type IIb hypoplasia and high angle mandible
occlusal plane. (Center) At age 6 years, with noted condylar head flattening and resorption. (Right) At age 10 years, with continued
condylar resorption changes and high angle mandible occlusal plane.
to move away from the skull base, there is an equal
and opposite force that is transmitted through to
the temporomandibular joint (Fig. 1). Condylar
resorption is a documented secondary effect of tra-
ditional mandible advancement operations,27–29 and
is thought to be secondary to avascular necrosis30 or
caused by genetic predilection such as in cases with
high plane angle mandibular hypoplasia. Histori-
cally, the gradual soft-tissue adaptation of distrac-
tion mitigated the risk of condylar changes.31 An
evidence-based review of condylar resorption fol-
lowing 6- to 10-mm movements reported a lower
(1.4 percent) rate after mandibular distraction
osteogenesis compared with sagittal split osteotomy
(6 percent).11 Because the recognized benefit of
mandibular distraction osteogenesis is to achieve
large or very early advancements, it is important to
assess critically condylar changes in these specific
situations to determine whether the risk is greater
than these reported rates after smaller movements
in adults.
Early dog studies by McCarthy demonstrated
flattening of the condylar head with cartilage
thinning, but with subsequent signs of repair.32 In
humans, they observed expansion of the affected
condyle in the first year after surgery, but consid-
ered it a favorable normalization.33 Recent small-
animal studies have demonstrated a discontinuity
of condylar cartilage after distraction of normal
mandible anatomy into an abnormal position,34
and more severe permanent changes in the pres-
ence of systemic factors such as immunosuppres-
sion.34 It is challenging to create an animal model
Copyright © 2020 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.
that represents the abnormal anatomy found in
the clinical population undergoing mandibular
distraction osteogenesis. There have been isolated
case reports of condylar resorption following
large-distance mandibular distraction osteogen-
esis,35 and a case series of 13 patients treated with
distraction for severe malocclusion reported con-
dylar resorption in 20 percent of cases, with a
superior and posterior displacement of the con-
dyle in the fossa.36 The degree of joint dysfunction
was associated with the magnitude of distraction
and the incidence of preoperative articular disk
displacement.
We must take these forces of mandibular dis-
traction osteogenesis into consideration when
we use this on infants in the first weeks of life for
the treatment of severe obstructive apnea associ-
ated with Pierre Robin sequence.37 [See Video 1
(online), which displays the critical considerations
in performing a neonatal mandible distraction
for airway improvement.] Researchers are exam-
ining the impact of these bilateral procedures on
the neonate condyle and mandibular arch. Com-
pared to adult procedures, neonate mandibular
distraction osteogenesis appears to change arch
form anterior to the osteotomy into a more nor-
mal parabolic shape, indicating the plasticity of
these young patients.38 The condyles, however,
appear to shift and rotate superolaterally, and
the long-term effects remain unknown.39 A ret-
rospective study of 10 Robin sequence patients
with a mean follow-up of 7 years, demonstrated
that mandibular length was shorter than in Robin
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 Plastic and Reconstructive Surgery • June 2020

sequence patients who did not undergo distrac-
tion, but that the mandibular ramus height was
comparable.40 Preoperative morphology was not
available, so presenting severity could not be con-
trolled, but the comparable ramal height suggests
that there was not an overall negative effect on
condylar growth.
Mandibular distraction osteogenesis long-
term studies are indicated. The literature does
suggest that some patients may be at increased
risk of progressive deterioration of the ramal-con-
dylar unit, whereas others likely have the poten-
tial to accommodate and resume normal growth.
Although the risk factors are not yet defined,
they are likely similar to those of traditional sur-
gery, such as intrinsic joint derangement and
high mandible plane angle. Mitigating interven-
tions that may be used in patients considered at
highest risk include unloading the condyle with
bone-anchored elastics,41 using a stabilizing plate
that transfers vertical pressure to the zygoma,42 or
using a halo-based device to avoid bidirectional
forces across the osteotomy (Fig. 2).42,43
ALVEOLUS
After alveolar distraction was introduced over
20 years ago, there was initial enthusiasm that it
would replace traditional grafting.44–46 This was
dampened with subsequent reports of patient
discomfort, technical challenges, increased risk
of complications, and the need for postoperative
patient compliance.47 A selective literature review
of alveolar distraction found that 85 percent of
the cases had minor complications that did not
interfere with treatment (primarily improper
inclination of vector, insufficient bone breadth,
or dehiscence), but 15 percent had major com-
plications (primary fracture of the basal bone,
distractor, or transport segment).48 The authors
concluded that the complexity of the technique
and the perceived higher complication risk limit
its popularity.48 There have been two randomized
clinical trials comparing alveolar distraction with
grafting.49,50 Bianchi et al. compared five patients
undergoing mandible alveolar distraction and six
patients undergoing grafting for implant place-
ment. They found comparable 2-year implant suc-
cess rates (94 and 95 percent), with greater bone
gain following distraction (10 mm versus 6 mm),
but more complications (60 percent versus 17
percent).49 Chiapasco et al. reported comparable
bone gain but lower remodeling, higher 3-year
implant success rate, and comparable complica-
tions in the distraction group compared to graft-
ing.50 The most recent meta-analysis reported a
greater mean gain with distraction (8 mm versus
4 mm) but a higher complication rate (47 percent
versus 24 percent).51 A recent systematic review
recognized distraction as a treatment for vertical

Fig. 2. Methods to decrease condylar pressure during distraction osteogenesis of the mandible
ramus. (Left) An L-shaped titanium plate (yellow area) fixated to the mandible coronoid can be
used to transmit force (arrows) directly to the zygoma instead of the temporomandibular joint
(Shakir S, Naran S, Lowe KM, Bartlett SP. Balancing distraction forces in the mandible: Newton’s
third law of distraction. Plast Reconstr Surg Glob Open 2018;6:e1856). (Right) Cranium-anchored
mandible distraction osteogenesis. The forces (arrows) are transmitted directly to the cranial foot-
plates of the distraction device (yellow area), bypassing the temporomandibular joint (Dong E,
Dempsey RF, Wirthlin JO, Buchanan EP. Cranial anchored mandible distraction osteogenesis. J Cra-
niofac Surg. 2019;30:e183–e186).

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 Volume 145, Number 6 • Craniofacial Distraction Osteogenesis

Fig. 3. Intraoral images of an adult patient with bilateral cleft lip and palate and absence of the premaxilla. (Above, left) Preopera-
tively, there was a 25-mm bone defect with a large oronasal fistula not amenable to traditional grafting. (Above, right) Bilateral
horizontal transport alveolar distraction was used to minimize the fistula and to create adjacent tooth-bearing segments. (Below,
left) Callus manipulation was performed early in the consolidation period to achieve the desired arch form, without the need for
additional grafting at the docking site. Orthodontic manipulation was then used for canine substitution into the central incisor
position. (Below, right) Final occlusion was achieved with prosthetic camouflage and by placing implants in the generated bone.

bone atrophy, but that more studies are required
to determine whether it is advantageous.52
Experts have used segmental transport alveo-
lar distraction as a salvage technique for large
defects not amenable to grafting, but it is tech-
nique sensitive and requires complex postopera-
tive management (Fig. 3). Transport distraction
of tooth-bearing segments with attached gingiva
offers a soft-tissue benefit over microvascular flaps
for these complex cases with planned implant
placement. Two-stage treatment of onlay bone
graft followed by vertical distraction53 or, in rare
cases, staged horizontal and vertical distraction54
can be used to successfully treat severely deficient
alveolar ridges. Considerations with these tech-
niques include the importance of careful vector
control, such as by resting the distractor against
adjacent teeth, and the need for temporary
anchorage devices to direct orthodontic molding
of the generated bone.55 Horizontal alveolar dis-
traction has also been successful in recreating the
maxillary and mandibular arch form after large
defects using horizontal transport distraction fol-
lowed by dynamic molding of the generated bone
or acute open callus manipulation and fixation
(Fig. 3).56–58 These complex staged approaches
can result in reconstructions that are comparable
to native anatomy.
Over the past 20 years of alveolar distraction,
the technique has not replaced more traditional
techniques of grafting59–61 or guided bone regen-
eration62,63 because of a higher rate of complica-
tions and technical challenge. These additional
considerations, however, are acceptable in severe
cases not amenable to traditional techniques.
MIDFACE
Le Fort I Distraction Surgery to Achieve Final
Occlusal Position
In a 2002 review, Shaw et al. suggested that
maxillary distraction might be superior to con-
ventional orthognathic surgery for cleft palate
hypoplasia but that, ethically, further studies
are required.64 A decade later, a similar review
reported that conventional Le Fort I surgery in
this population had high horizontal and vertical
relapse,65 but that the same or larger movements
with distraction osteogenesis had good stability.66
In a recent critical Cochrane Library Systematic
Review, only six articles from a single center met
the criteria of a randomized controlled study of

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these two approaches with appropriate outcome
measures.67–72 The conclusions were that distrac-
tion provided greater advancement and stability,
no additional patient distress or difference in
speech outcome, but greater long-term patient
satisfaction compared with conventional Le Fort
I distraction73 (Reference 73 Level of Evidence:
Therapeutic, II). Because of additional cost, Le
Fort I distraction has not replaced conventional
Le Fort I for small occlusal changes in the normal
population, but has an established role in large
advancements such as the cleft palate popula-
tion. Le Fort I distraction for large movements in
the early teenage years can serve as the definitive
orthognathic treatment with appropriate presur-
gical planning of overcorrection to anticipate pro-
jected mandible growth.
Segmental and Rotational Subcranial Movement
Surgical pioneers developed the subcranial
osteotomy into the standard of care for treat-
ing severe midface hypoplasia.74–78 With the rec-
ognition of the need for differential movement
of parts of the Le Fort III segment to achieve
facial harmony, Obwegeser advocated the con-
cept of combined segmental Le Fort osteotomies
with differential mobilization in 1969.79 Polley
and Figueroa revisited this differential concept
through tiered midface osteotomies in a “pig-
gyback” fashion for simultaneous correction of
orbital, midface, and occlusal abnormalities.80
Soft-tissue resistance limited the degree of tradi-
tional Le Fort III advancement and contributed to
relapse. The associated lack of anterior maxillary
growth following surgery implied that definitive
orthognathic correction in early adulthood was
still required.81–83
Development of midface distraction in the
early 1990s had the reported benefits of greater
advancement, improved stability, lower relapse,
and superior airway outcomes.5,84–87 With these
advantages, single-piece Le Fort III distraction
largely replaced traditional subcranial advance-
ments in adolescents.88 As with traditional sur-
gery, there is now a resurgent awareness of the
shortcomings of en bloc movement in addressing
multilevel asymmetries, leading to approaches
that combine the benefits of distraction with the
directional control of traditional techniques.
Combined distraction of Le Fort III and Le
Fort I osteotomies have been described for the
treatment of syndromic midface hypoplasia to
differentially correct orbital and occlusal hypo-
plasia.89–92 Other refinements include the use of
nasal passenger grafts, cerclage hinge control for
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Plastic and Reconstructive Surgery • June 2020
rotational movements, and modifications of Le
Fort III segmental osteotomies.93 Appreciation of
the distinct facial ratios of different syndromes
has led to techniques such as the Le Fort II dis-
traction with zygomatic repositioning to correct
greater central vertical and sagittal deficiency
that normalizes facial ratios in conditions such as
Apert and Pfeiffer syndromes and achondropla-
sia (Fig. 4).93–95 (Reference 94 Level of Evidence:
Therapeutic, III). A recent review of a consecutive
midface distraction series showed that transfusion
and operative times were greater for Le Fort II
distraction with zygomatic repositioning than for
Le Fort III; however, controlling for patient diag-
nosis and concomitant procedures, there were no
significant differences in complications between
these two procedures.96 The bipartition mono­
bloc distraction technique has similarly combined
traditional osteotomies and wire fixation with the
power of distraction to achieve favorable differen-
tial changes in these complex cases, but does have
an increased complication rate compared with Le
Fort III advancements alone.97
Relatively small (<10 degrees) counterclock-
wise rotation of Le Fort I osteotomies corrects high
plane angle occlusal deformities and increases
posterior airway dimensions.98–100 Tulasne and
Tessier recognized the importance of this in 1986
when they proposed the subcranial “Procédure
Integral” to correct the rotation deformity of
Treacher Collins syndrome.101 Adoption of this
technique failed because of relapse of mandibu-
lar position, the need for extensive grafting, insta-
bility of the maxillary occlusal rotation, and high
soft-tissue resistance.101,102 Three decades later,
Tessier’s vision of a subcranial movement was
achieved through craniofacial counterclockwise
distraction osteogenesis. [See Video 2 (online),
which displays the concept of subcranial rotation
distraction including expected airway changes.]
This technique has been successful for decannu-
lation of tracheostomy-dependent patients with
Treacher Collins syndrome and those with auricu-
locondylar syndrome and craniofacial microsomia
who were resistant to isolated linear distraction
techniques103 (Reference 103 Level of Evidence:
Therapeutic, IV).
Subcranial distraction continues to evolve by
combining the classic concepts of segmental oste-
otomies to achieve differential movements with
the power of gradual distraction to achieve move-
ments and vectors not previously possible. The
advantages achieved through this combination
will maintain distraction as the standard of prac-
tice in severe midface hypoplasia.
 Volume 145, Number 6 • Craniofacial Distraction Osteogenesis

Fig. 4. (Left) The dysmorphology of Apert syndrome requires differential segmental movement of the forehead (blue), zygo-
mas (yellow), and maxilla (orange) to normalize facial ratios. (Right) The Le Fort II distraction with simultaneous zygomatic
repositioning procedure along with a custom forehead implant achieves this goal through the combination of distraction
and traditional fixation techniques (Hopper RA, Kapadia H, Morton T. Normalizing facial ratios in Apert syndrome patients
with Le Fort II midface distraction and simultaneous zygomatic repositioning. Plast Reconstr Surg. 2013;132:129–140).

CRANIUM been shown to have a secondary beneficial effect

Intracranial Effects of Posterior Cranial Vault
Distraction
After early frontal bone advancement resulted
in unacceptable relapse, posterior cranial vault
expansion became a first treatment for multiple
suture synostoses.104 Removal of the posterior cra-
nium for remodeling required dissection over the
confluence of the sagittal and transverse venous
sinuses (torcula), with the potential risk of life-
threatening bleeding.105,106 When the Birmingham
craniofacial group first described posterior vault
distraction osteogenesis in 2009, it had relatively
high rates of minor complications, but through
refinement, it has become a popular method for
early expansion.107–109 Advantages over remodeling
include avoidance of epidural dissection over the
torcula that maintains vascularity of the bone flap,
gradual volume expansion to prevent dead space,
and stabilization of the expansion during healing by
the spanning devices. [See Video 3 (online), which
displays the critical considerations in performing
posterior cranial vault distraction including vector
alignment and subcranial decompression.]
Posterior vault distraction provides more vol-
ume increase per millimeter of movement than
a fronto-orbital advancement (Fig. 5),110 and has
on frontal bone morphology.111 Early expansion
of the posterior vault can thus postpone the need
for fronto-orbital advancement until after the first
year of life to minimize the need for secondary
surgery.107–109,111 An early comparison of posterior
vault distraction osteogenesis with conventional
posterior osteotomy did not find any difference
in perioperative safety and morbidity profiles,112
but given the relatively low risk of serious com-
plications in elective cranial surgery,113 much
larger numbers would be needed to detect a pos-
sible difference. A literature review from 2009 to
2013 included 86 patients, and found an overall
30 percent complication rate, with the most com-
mon occurrences being cerebrospinal fluid leak
(10 percent), local infection or dehiscence (7
percent), and device loosening (6 percent).114 A
more recent review of the National Surgical Qual-
ity Improvement Program from 2012 to 2016
included 94 posterior vault distraction osteogen-
esis cases and found no reoperations, readmis-
sions, or serious events within 30 days of surgery.115
Increased age at surgery was the only predictor of
increased operating time and transfusion rate.
It is unclear whether posterior vault distrac-
tion osteogenesis is true distraction osteogen-
esis, because the cross-section of bone across the

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 Plastic and Reconstructive Surgery • June 2020

Fig. 5. Schematic depictions of a skull with turribrachycephaly showing differential volume

expansion of posterior (blue) versus anterior (orange) cranial expansion. Because of the larger sur-
face area of the posterior cranial transport segment, the volume expansion has been estimated at
5.8 cm2/mm advancement with posterior distraction compared to 4.6 cm2/mm with fronto-orbital
advancement.

osteotomy is thin, and there has been no docu-
mented formation of the typical regenerate tis-
sue.116 Healing of the expanded osteotomy may
result instead from the osteogenic potential of
the neonatal dura.117 What has become apparent,
however, is that there is a secondary beneficial
effect of distraction on structural changes in the
posterior fossa contents and resolution of Chiari
malformation. Chronic cerebellar tonsillar her-
niation (Chiari I malformation) is commonly
found in syndromic craniosynostosis and nonsyn-
dromic lambdoid synostosis,118,119 and traditional
treatment of the malformation involves direct
suboccipital decompression at the foramen
magnum.120 Several centers have demonstrated
improvement or resolution of an existing Chiari
malformation following posterior cranial distrac-
tion for multisuture synostosis (Fig. 6)121–124 (Ref-
erence 123 Level of Evidence: Therapeutic, IV).
A suboccipital decompression may also be incor-
porated into the design of a posterior distraction
osteotomy for further decompression.125 The
Birmingham craniofacial group has also exam-
ined posterior vault distraction osteogenesis as
a primary treatment of Chiari malformation in
five patients aged 2 to 8 years.126 All had symp-
tomatic resolution, and the three with syrinxes
had reduction in size. Longer follow-up of these
patients is underway.
Anterior Cranial Vault Distraction
Although posterior vault distraction osteo-
genesis has been more widely accepted, the role
of anterior cranial vault distraction remains more
controversial. Sugawara et al. in 1998 described dis-
traction of a fronto-orbital advancement osteotomy,
and there have been additional modifications over
time (Fig. 7).127–129 Other centers have described this
technique for bilateral coronal or complex multisu-
ture craniosynostosis.130–133 Variations in technique
include simple suturectomy,134 one-piece bilateral
frontal bone with superior orbital rims,135 hemi-
frontal craniotomy with en bloc bandeau,136–138 and
complete fronto-orbital remodeling (Fig. 7).129 Pro-
ponents of anterior cranial distraction hypothesize
that the gradual movement of the combined dura-
bone unit will create a more supportive change
in the brain to decrease relapse and the need for
secondary surgery. This is in contrast to one-stage
advancement and remodeling, which acutely cre-
ates space for the brain secondarily to expand.
Fearon et al. published a study comparing tra-
ditional frontal calvarial advancement to frontal
bone distraction in patients with Apert syndrome.139
With equal advancement in both groups, they found
that there was no significant advantage to distrac-
tion regarding blood loss or anthropometrics. More
operations were required in the distraction group,
and some of these patients had unintended bone

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 Volume 145, Number 6 • Craniofacial Distraction Osteogenesis
Fig. 6. (Left) Magnetic resonance imaging scan of a patient with multisuture synostosis and Chiari type
I malformation (shaded yellow, top arrow) with associated syrinx (bottom arrow). (Right) After posterior
vault distraction, the Chiari malformation and syrinx have resolved.
deposition. A comparison of six traditional advance-
ments with six distraction patients noted no differ-
ence in aesthetic outcomes or blood loss between
groups, but noted more new-onset strabismus in the
traditional group.140 Corkum et al. reported a system-
atic literature review including 223 one-stage and 69
distraction operations for unilateral coronal synos-
tosis, and found a nonsignificant trend toward early
favorable Whitaker scores with distraction, but not
other differences.141 Mundinger et al.142 performed
a review of all comparative studies between cranial
distraction and one-stage surgery, and also found
no significant differences. Anterior cranial surgery
holds promise but, at this time, has not demon-
strated a clear superiority over single-stage surgery.
Further evolution of the technique and more long-
term comparative studies will help clarify its future
role and to justify the additional costs and need for
device removal.
Monobloc Distraction versus Staged Cranial and
Subcranial Advancements
Traditional monobloc advancement had a high
rate of serious infections from the acute dead space
created that communicated the anterior fossa with
the sinuses,143,144 and was reserved for the most severe
dysmorphology.145 Application of distraction osteo-
genesis to the monobloc osteotomy allowed the fron-
tal lobe to advance gradually with the bone, resulting
in marked decreases in infectious complications and
relapse, and making this form of advancement the
new standard145,146 (Reference 146 Level of Evidence:
Therapeutic, III). [See Video 4 (online), which
displays the critical considerations in minimizing
Copyright © 2020 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.
complications during early monobloc distraction
advancement.] This increased safety profile of
monobloc distraction has created a debate regarding
whether its role should expand so that early fronto-
facial surgery replaces staged fronto-orbital advance-
ment and adolescent midface surgery as the new
standard of treating syndromic conditions.
Advocates of early monobloc distraction recog-
nize the attraction of simultaneous correction of
brachycephaly, exorbitism, airway obstruction, and
malocclusion. Arnaud and Di Rocco reported the
largest series to date of 104 cases, with a 28 percent
complication rate, including 20 percent cerebrospi-
nal fluid leak.145 Occlusal correction occurred in 77
percent, and airway resolution occurred in all with
obstruction. Advocates of a staged fronto-orbital
advancement and later midface approach to these
patients purport a higher safety profile and the
opportunity for differential frontofacial advance-
ment. Orbital correction and airway improvement
is achievable by either technique, but the lateral
orbital rim advancement following monobloc dis-
traction is considered superior to Le Fort III.147–149
Sagittal growth potential after both procedures is
similarly blunted, emphasizing the need for over-
correction, which may be more challenging with
the monobloc algorithm because the midface is
treated earlier than with the staged approach.150–152
Lack of growth after early monobloc distraction
may predispose to a higher rate of complicated sec-
ondary midface operations later in life compared to
delaying subcranial surgery until adolescence.153,154
It is accepted that the complication rate is
greater following monobloc distraction compared
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Fig. 7. Two-year-old patient with Apert syndrome and brachycephaly from multisuture fusion. (Below)
The orbital bandeau and frontal bone has been removed and modified, as in a standard fronto-orbital
advancement, but was fixated back against the dura with bilateral internal distraction devices. (Above,
left) The devices were activated after 4 days’ latency at 1 mm/day until desired brow position was
achieved at 20-mm advancement. (Above, right) The devices were removed at 12 weeks, with healing
of the distraction gap. The skull was examined at age 8 during a midface procedure and demonstrated
complete ossification.

with Le Fort III distraction. A systematic review
showed a 33.7 percent incidence of major compli-
cations (including 20 percent cerebrospinal fluid
leak) with monobloc distraction versus 17.6 percent
incidence for LeFort III distraction.155 A retrospec-
tive comparison between the two techniques at one
center found that monobloc distraction was 5.8
times more likely to have complications, and that
infections occurring after monobloc distraction
were more likely to be serious and involve the cra-
nial cavity.156
Despite these studies, the lack of randomized
or controlled studies challenges the comparison
of these two approaches, as does the need for
outcomes over the first decade of growth. The
critical question is whether the periorbital and
single-stage multilevel benefits of monobloc dis-
traction outweigh the increased safety profile and
opportunity for differential overcorrection of a
staged approach (Fig. 8). Because a randomized
trial in unlikely, the answer will require a trans-
parent comparison of long-term outcomes in a
similar population. Perhaps we should not debate
whether a center should be performing one
approach to the exclusion of another, but instead
examine what the expected distribution of each
approach is within a center that tailors treatment
to presenting dysmorphology.154
SUMMARY
Over the past 30 years, we have explored
the potential of distraction osteogenesis in the

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Copyright © 2020 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.
 Volume 145, Number 6 • Craniofacial Distraction Osteogenesis

Fig. 8. Computed tomographic scans with colorized globes of two patients with Pfeiffer syndrome who
underwent different treatment pathways at our center. Patient 1, at age 9 months (above, left); at age 18
months after fronto-orbital advancement (above, center); and at age 7 years, immediately after Le Fort III
distraction (above, right). Patient 2, at age 8 months (below, left); at age 16 months, immediately after early
monobloc distraction (below, center); and at age 6 years (below, right). Both patients had preceding posterior
vault distraction at 6 months. Patient 2 underwent early monobloc distraction because of prolapsing exoph-
thalmos and severe nasopharyngeal compression.

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Copyright © 2020 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.

craniofacial skeleton. It may not have brought a
“farewell to fixation,” but what traditional tech-
niques has it replaced? Based on the existing liter-
ature, it is clear that distraction can achieve large
mandible movements but that it has not overcome
the challenges of high occlusal plane angle, condy-
lar resorption, or neurosensory changes. Sagittal
split osteotomy remains the standard for precise
final occlusal changes. Alveolar distraction is a pow-
erful and evolving technique. Its higher complica-
tion rate and technical difficulty have prevented
it from replacing traditional grafting for implant
placement, but it shows promise in restoring a
native environment in cases with severe defects
using a staged approach. Subcranial techniques are
combining the power of distraction with a revival of
traditional osteotomies to achieve novel differential
movement. Subcranial and monobloc distraction
have replaced traditional advancement because of
the ability to achieve safer, larger, and more stable
advancements. More long-term outcome studies
of comparable populations treated with mono-
bloc approach versus a staged approach will help
us tailor the appropriate treatment for syndromic
patients. We have yet to demonstrate an improved
safety potential for posterior cranial distraction, but
its ability to achieve large cranial volume increases
with favorable intracranial changes has made it a
standard first-stage treatment in many centers. The
potential of anterior cranial distraction to achieve
more efficient, stable results continues to be
explored, but long-term comparisons of shape and
function will be required before it can compete with
the standard of fronto-orbital advancement. The
first three decades of distraction have been an era
of exploration, whereas the road ahead will be one
of quantitative validation and careful evaluation.
The evolution of craniofacial distraction continues.
Richard A. Hopper, M.D.
Craniofacial Center
Seattle Children’s Hospital
4800 Sand Point Way NE
Seattle, Wash. 98105
richard.hopper@seattlechildrens.org
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