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8/13/2019 Craniofacial Distraction, A Review
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Int. J. Oral Maxillofac. Surg. 2001; 30: 89103
doi:10.1054/ijom.2000.0033, available online at http://www.idealibrary.com on
Invited review
Craniofacial distractionosteogenesis: a review of theliterature. Part 1: clinicalstudiesG. Swennen, H. Schliephake, R. Dempf, H. Schierle, C. Malevez: Craniofacial
distraction osteogenesis: a review of the literature. Int. J. Oral Maxillofac. Surg.
2001; 30: 89103. 2001 International Association of Oral and Maxillofacial
Surgeons
Abstract.A review of the literature dealing with distraction osteogenesis (DO) of
the craniofacial skeleton, provided by a PUBMED search (National Library of
Medicine, NCBI; revised 3 April 2000) from 1966 to December 1999 was
conducted. Key words used in the search were distraction, lengthening, mandible,
mandibular, maxilla, maxillary, midface, midfacial, monobloc, cranial, craniofacial
and maxillofacial. This search revealed 285 articles. One hundred and nine articles
were clinically orientated and were analysed in detail in this study. The type of
distraction, indications, age, type of surgery, distraction rates and rhythms, latency
and contention periods, amount of lengthening, follow-up period, relapse,complications and the nature of the distraction device were analysed. This review
revealed that 828 patients underwent DO of the craniofacial skeleton; 579
underwent mandibular DO, 129 maxillary DO, 24 simultaneous mandibular and
maxillary DO and 96 midfacial and/or cranial DO. Craniofacial DO has proven to
be a major advance for the treatment of numerous congenital and acquired
craniofacial deformities. Treatment protocols and success criteria for craniofacialDO are suggested on the basis of these results. There is still, however, a lack of
sufficient data, especially on follow-up and relapse, so that treatment strategieshave to be validated by long-term studies in the future.
Gwen Swennen1,2,Henning Schliephake1,Rupert Dempf1, Hannes Schierle1,3,Chantal Malevez2
1Department of Oral and Maxillofacial Surgery,Medizinische Hochschule Hannover,Hannover, Germany2Department of Oral and Maxillofacial Surgery,Childrens Hospital, University of Brussels,Belgium3Private Practice for Oral and MaxillofacialSurgery, Karlsruhe, Germany
Key words: distraction; mandible; maxilla;midface; cranial; treatment protocols; successcriteria.
Accepted for publication 20 November 2000
Distraction osteogenesis (DO) is the pro-cess of generating new bone in a gapbetween two bone segments in response
to the application of graduated tensilestress across the bone gap. The tech-
nique of bone lengthening by DO wasfirst described in 1905 by C13,when he reported lengthening of a
femur byaxial distraction forces. Otherreports1,2 also mention the concept of
bone lengthening in the tibia, but itremained undeveloped until Dr GavrielA. I5456, a Russian physician,
further developed the technique in the
1950s in Kurgan in West Siberia. Formore than 35 years he successfullyapplied the technique of DO to the
enchondral bone of the upper and lowerextremities. A unique feature of the
distraction technique is that bone regen-eration by DO is accompanied by simul-taneous expansion of the functional soft
tissue matrix, including blood vessels,nerves, muscles, skin, mucosa, fascia,
ligaments, cartilage and periosteum.These adaptive changes of the surround-ing soft tissues through the tension that
is generated by the distraction forces
applied onthebone, is called distractionhistogenesis22. Distraction osteogenesisseemed to be a promising new method in
the reconstruction of the membranousbones of the human craniofacial skel-
eton, when in 1992 MC et al.83
reported the first clinical application inthe Western literature of mandibular
lengthening by gradual distraction inpatients with hemifacial microsomia and
Nagers syndrome. Since then there hasbeen an explosion of reports in the lit-erature on DO in the craniofacial skel-
eton. In recent years, DO has become
0901-5027/01/020089+ 15 $35.00/0 2001 International Association of Oral and Maxillofacial Surgeons
8/13/2019 Craniofacial Distraction, A Review
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increasingly popular and opened newtherapeutic perspectives for the treat-
ment of numerous congenital andacquired craniofacial skeletal anomalies.
Different treatment protocols, however,are applied by different groups. Despitethe growing body of literature on the
applications of craniofacial DO, import-ant parameters such as age, rhythm,
rate, latency period, contention periodare not rigidly established. Excellent
comprehensive reviews have been pub-lished4,22,24,82,133,134,137,145, however,
none of these provide an analysis ofthese parameters in detail. Therefore, theliterature dealing with DO of the cranio-
facial skeleton was reviewed comprehen-sively. The purpose of this review wastwo-fold: (1) to evaluate clinical indi-
cations and DO parameters by compar-ing different treatment protocols and
results obtained by different craniofacialteams; (2) to suggest treatment protocols
and success criteria for craniofacial DObased on clinical, experimental andscientific investigations to help provide
more objective data in the future.
Materials and methods
A review of the literature on DO of the
craniofacial skeleton, provided by aPUBMED search (National Library of
medicine, NCBI, New Pubmed System;revised 3 april 2000), was conductedfrom 1966 to December 1999. Key words
applied in the search were distraction,lengthening, mandible, mandibular,
maxilla, maxillary, midface, midfacial,monobloc, cranial, craniofacial and
maxillofacial. This initial search revealed285 articles. An additional three articles
relating to the subject were submitted tothe study. Of this total study sample of288 articles, 109 were clinical and 99
were experimental articles; 54 articleswere primarily on a scientific basis andwere excluded because they provided no
sufficient data; of the remaining articles,11 were excluded due to lack of appro-
priate data and 15 because they werewritten in languages, for which trans-
lation was not available (12 in Russian,two in Chinese and one in Hebrew)(Table 1). The study sample of this
review consisted of 109 articles that were
analysed in detail. Flow sheets weremade of each article with the specificparameters relative to DO. The author,
type of distraction, indications, numberof patients, age, type of surgery, distrac-
tion rates and rhythms, latency and con-tention periods, amount of lengthening,
follow-up period, relapse, complicationsand the nature of the device wererecorded for each article on the flow
sheets and analysed. The articles weredivided into four major groups accord-
ing to the site of distraction: mandible,maxilla, combined mandible and max-illa, and midface/cranium. Although
some authors have published results ofidentical patients in more publications,
this could not be verified in detail.For analysis, the data of all different
publications were used.
Results
A total of 109 articles were analysed; 74
(67.9%) were related to mandibular DO,16 to maxillary DO (14.7%), three to
simultaneous mandibular/maxillary DO(2.8%) and 23 to midfacial and/orcranial DO (21.1%); seven of these
articles were related to two or moreprocedures (Table 2). Indications for
craniofacial DO are shown in Table 3.
Mandibular distraction osteogenesis
A total of 579 patients (70.0%) under-went distraction procedures involving
the mandible(Table 3). These mandibu-lar DO procedures were divided intofour groups. Of the 579 patients, 430
(74.3%) underwent mandibular length-ening (Group 1), 38 patients (6.6%)
mandibular widening (Group 2), 16patients (2.8%) alveolar reconstruction(Group 3) and 95 patients (16.4%) bone
transport and compression DO (Group4). Results of mandibular DO par-
ameters for each group are shown inTable 4.
Group 1: Mandibular lengthening
Of the 430 patients that underwentmandibular lengthening through DO,
345 (80.2%) had congenital micro-
gnathia8,11,14,16,19,2331,42,45,46,51,52,53,57,
59,62,63,6567,72,73,79,8385,87,88,94,98,101,102,
105,107,108,115,120,123,126,130,135,141,144 45
(10.5%) had acquired micro-
gnathia8,25,29,30,31,36,59,6264,66,84,88,91,98,
108,119,130,141,147 and 40(9.3%) had man-
dibular retrognathia26,31,91. Detailed
data regarding age were available in 156
patients (45.2%) with congenital micro-
gnathia and in 26 patients (57.8%) with
acquired micrognathia. In the congenital
micrognathia sample, age varied from 6
days to 19 years and most of the patientswere distracted in the age groups, 26
and 712 years. Patients with acquired
micrognathia were distracted at an age
varying from 1.5 to 64 years with most
of the patients distracted in the 712
years age group. Data regarding age
were available in 37 patients (92.5%)
with mandibular retrognathia; all
patients were distracted between the age
of 15 and 20 years. A total of 289
patients (83.8%) with congenital micro-
gnathia, 40 patients (88.9%) withacquired micrognathia and 40 patients
(100%) with retrognathia had infor-mation regarding the surgical technique.A complete osteotomy was performed in
166 patients (57.4%) with congenitalmicrognathia, while 123 (42.6%) under-
went a corticotomy. In the acquiredmicrognathia sample a total of 23patients (56.1%) underwent an osteo-
tomy, while 18 patients (43.9%) acorticotomy. A body osteotomy was per-
formed in all patients with mandibularretrognathia. Data regarding the distrac-tion rate were available in 300 patients
(86.9%) with congenital micrognathia,
37 (82.2%) with acquired micrognathiaand 40 (100%) with mandibular retrog-nathia. The distraction rate of 1 mm
daily was the most frequent for eachgroup. Data on distraction rhythm,varying from two to four times daily,
were reported in only 174 patients (58%)in the congenital micrognathia sample
and in 10 patients (27%) in the acquiredmicrognathia sample. All patients in theretrognathia sample had data on distrac-
tion rhythm, ranging from three times0.33 mm to twice 0.5 mm. A total of 325
Table 1. Articles on distraction osteogenesisof the craniofacial skeleton
Clinical DO 109Experimental DO 99Scientific DO 54Not relevant 11Other language 15Total 288
Table 2. Articles on clinical distraction osteogenesis
Mandibular DO 74 74Maxillary DO 16 4* 12Simultaneous mandibular/maxillary DO 3 3Midfacial and/or cranial DO 23 3** 20Total 109
*Of the 16 articles on maxillary DO, four concerned also mandibular DO.**Of the 23 articles on midfacial and/or cranial DO, three concerned also mandibular DO.
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Table 3. Indications for distraction osteogenesis of the craniofacial skeleton
Number % %
Mandibular DOHemifacial microsomia (HFM)28,29,30,31,46,53,59,67,73,65,66,79,83,87,88,101,102,105,123,126,130,135 198 34.2Segmentalbone defect (trauma, tumor)6,9,76,113,116118,121,122 90 15.5Class II26,31,91 40 6.9Acquired micrognathia(trauma,TMJ ankylosis)8,25,29,30,31,59,64,65,66,85,88,97,98,108,141 39 6.7Transverse discrepancy41,60,142,143 35 6.0Craniofacial microsomia (CFM)11,13,14,84,85,98,108,130 32 5.5
Treacher-Collins Syndrome (TC)8,29,72,65,66,87,88,94,98,108,144
27 4.7Congenital micrognathia8,42,45,51,59,84,87,98,105 24 4.1Obstructive SleepApnoe Syndrome (OSAS)16,52,91 18 3.1Alveolar deficiency37,38,47,69,139 16 2.8Nager Syndrome59,79,83,87,108,115,144 10 1.7Pierre Robin Syndrome8,57,58,88,107,120 8 1.4Goldenhar Syndrome8,11,87,98,108 6 1.0Fibula36,119,147 6 1.0Arthrogryposis11,98 3 0.5Hypoglossia-hypodactyly Syndrome8,99 2 0.4Hanhart Syndrome29,73 2 0.4Post irradiation therapy8 2 0.4Condylar resorbtion124 2 0.4Moebius Syndrome62,63 2 0.4Histiocytose X121,122 2 0.4Rubinstein-Taybi Syndrome79 1 0.2Cerebro Costomandibular Syndrome79 1
Tessier cleft VII65
1Retrognathia/facial cleft65 1Plagiocephaly23 1Kampomele dysplasie115 1Trisomie 18115 1Morpheaform sleroderma23 1Amnionband Syndrome8 1Neurofibromatosis8 1Glossoptosis-micrognathia107 1Aglossia-micrognathia8 1Silver-RussellSyndrome61 1Facial skoliosis58 1Bilateral dysostosis mandibularis141 1Subtotal 579 100 70.0Maxillary DOOrofacial clefts 115 89.2
Unilateral cleft lip-palate (UCLP)3,34,70,89,106,127,128,141 55
Bilateral cleft lip-palate (BLCP)34,70,89,103,106,127,128,132,146
34Undefined clefts104 18Unilateral cleftpalate (UCP)70,89 8
Alveolar defect37 5 3.9NasomaxillaryDysplasia89 2 1.6Prognathism89 2 1.6Alveolar atrophy37,47 2 1.6Cleidocranial Dysostosis19 1 0.8Craniosynostosis (CST)70 1 0.8Oral-acral syndrome32 1 0.8Subtotal 129 100 15.6Simultaneous mandibular/maxillary DOHFM90,95,96 23 95.8TC96 1 4.2Subtotal 24 100 2.9Midfacial and/or cranial DOCraniosynostosis (CST) 86 90.0
Crouzons S5,10,12,19,20,21,27,68,125
36Aperts S5,10,12,18,19,20,27,77 19UndefinedCraniosynostosis77,81 15Pfeiffers S7,10,11,12,17,18,19,21,100 12Unilateral Coronal Synostosis71 1Bilateral CoronalSynostosis49 1Sagittal Synostosis125 1CarpentersS131 1
Midfacial cleft10,12 3 3.1UCLP15,19 3 3.1Unilateral CFM14,19 2 2.1BCLP/severe maxillary atrophy48 1 1.0Rare cleft19 1 1.0Subtotal 96 100 11.6
Total 828 100
Craniofacial distraction 91
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respectively; only 62 patients (26.2%)were followed-up for 1 year or longer and
only six relapses (4.0%) were reported. Atotal of 26 patients (57.8%) in the ac-
quired micrognathia sample had data onfollow-up varying from 3 months to 4years; only nine patients (34.6%) were
followed-up for 1 year or longer. Data onrelapse were only available in 10 patients
(22.2%); none of these patients showedskeletal relapse. No data regardingfollow-up and relapse were reported in
the mandibular retrognathia sample.Data on complications were available for
311 patients (72.3%); a total of 86 compli-cations were reported(Table 5).
Based on these results, the commonstandard appears to be a distractionrate of 1 mm daily, a latency period of 57
days and a contention period of 68weeks for patients with congenital or ac-
quired micrognathia and retrognathiaundergoing mandibular lengthening. In
patients with congenital or acquiredmicrognathia, both surgical techniques(complete osteotomy/corticotomy) can be
performed and both devices (external orinternal) can be applied. In retrognatic
patients a complete osteotomy of themandibular body and bone-borne inter-nal devices are recommended(Table 6).
Group 2: Mandibular widening
A total of 38 patients underwent sym-physeal widening through DO41,60,61,98,
99,142,143. Detailed information on agewas given in 27 patients (71.1%); nine
patients (33.3%) were distracted between13 and 16 years, 14 patients (51.9%)older than 16 years and four patients
(14.8%) younger than 13 years. Further-more, mandibular widening was
reported in 10 patients with unspecifiedage between 13 and 31 years41. Thus, a
total of 33 patients (89.2%) were dis-tracted at an age older than 12 years andonly four patients (10.1%) at an age
between 212 years. Three of these fourpatients were syndromic cases. In all
patients a midline osteotomy was per-formed. A total of 12 patients were dis-tracted at 1 mm daily, while 26 were
distracted at less than 1 mm daily; 24were distracted at 0.75 mm daily (three
times 0.25 mm), one at 0.5 mm andanother at 0.33 mm. Latency periods
were reported in all patients. The 57day latency period was the most com-
mon. Data regarding the contentionperiod were given in 22 patients (57.8%).The 68 weeks contention period was
reported most frequently. A total of 23patients (60.5%) had data on the amountof symphyseal widening varying from
3.9 to 14 mm. Only 16 patients (42.1%)had follow-up data varying from 6 to
45 months and none of all patients hadinformation on relapse. Of the 38
patients that underwent mandibularwidening through DO, two (5.3%) hadexternal devices and 36 (94.7%) inter-
nal monodirectional devices; 33 weretooth-borne and three bone-borne
devices. Data on complications werereported in 37 patients (97.4%); a total
of 10 complications (29.7%) occurred(Table 5).
Thus, it is recommended to perform
mandibular widening from the age of 12years after a complete symphyseal
osteotomy using an internal device, adistraction rate ranging from 0.75 to1 mm daily, a 57 day latency period and
a 68 weeks contention period(Table 6).
Group 3: Alveolar reconstruction
In this group 16 patients underwentalveolar reconstruction through vertical
DO11,37,38,47,69,139. Data regarding agewere available in 15 patients (93.8%) andvaried from 17 to 69 years. A segmental
osteotomy was performed in all patients.
A total of 10 patients had a distractionrate of 1 mm, while six had a rate of0.5 mm. Data regarding distraction
rhythm were available for only ninepatients (56.3%); eight patients had arhythm of 0.5 mm twice a day while one
0.5 mm once daily. The 57 days latencyperiod and the 8 week contention period
were the most common. In group 3 alldevices that were used were intraoraldevices; nine patients (56.3%) had inter-
nal monodirectional devices and sevenpatients (43.8%) had implant-borne
Table 5. Complications of distraction osteogenesis of the craniofacial skeleton
Distraction procedure
Total %Length
Mandibular
Bone TMaxill.adv/alv
Mand/maxill.
Midf/cranialWiden. Alveol.
Complications
Mech. problems (pin loosening/accidental trauma) 34 1 1 1* 7 44 33.1Transient hypesthesia inferior alveolar nerve 15 15 11.3
Minor local infection 5 4 1 2 12 9.0Pin infection 5 1 1 4 11 8.3Premature consolidation 5 4 1 10 7.6Transient weakness r.marginalis facial nerve 9 9 6.7Limited skeletal advancement 6 6 4.6Hypertrophic scar 4 4 3.0Asymmetric advancement 3 3 2.3Ankylosis (zygoma-coronoid processus) 3 3 2.3Severe infection 3** 3 2.3Tooth damage 1 2 3 2.3Tooth mobility 2 2 1.5Incomplete osteotomy 2 2 1.5Loss of distraction implant 1 1 2 1.5Incorrect distraction vector 1 1 0.8Pseudarthrosis 1 1 0.8Haematoma 1 1 0.8
Periodontal pocket 1 1 0.886 10 1 7 3 1 25 133 100
*Loosening of IMF; **2 meningitis, 1 H.flu.pneumonia.
Craniofacial distraction 93
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devices; six of these were used for pros-
thetic treatment after distraction wascompleted37,38, while one was removed
at the end of the activation period69.Data on the amount of lengthening werereported in 15 patients (93.8%) and varied
from 4 to 15 mm. Internal bone-bornedevices provided greater lengthening
(715 mm) compared to implant-borne
devices (4
8 mm). Follow-up was men-tioned in 14 cases (87.5%) and rangedfrom 1 to 13 months. None of these ar-ticles provided data on skeletal relapse.
Data on complications were only avail-able in six patients; only one complication
occurred (Table 5).It can be recommended to perform
mandibular alveolar reconstruction from
the age of 16 years after a segmentalosteotomy using a distraction rate of
1 mm daily, a latency period of 57 daysand a contention period of 8 weeks withan internal bone-borne device. A 0.5 mm
rate and a 46 months contention periodis recommended when a distraction
implant is used, that will serve for pros-thetic treatment after the contentionperiod(Table 6).
Group 4: Bone transport
Bone transport DO was performed in a
total of 50 patients. Six patients (12%)underwent bifocal bone transport DOfor reconstructionof a neocondyle (TMJ
sample)9,121,122,124, while 42 patients(84%) underwent bifocal bone trans-
port6,9,76,117,118 and 2 patients (4%) tri-
focal bone transport76,113 DO for
reconstruction of segmental defects due
to trauma or tumor resection. A total of
45 patients underwent reconstruction of
segmental defects through compression
DO116118. Detailed data on age were
reported in 41 patients (82%) who
underwent bone transport DO. A total
of 39 patients (95.1%) were distractedabove the age of 16 years, varying from
20 to 68 years. Data on age were only
available in seven of the patients (15.6%)
who underwent compression DO; they
all were distracted at an age varying
from 20 to 35 years. A reverse-L oste-
otomy was performed in all six patients
who had TMJ reconstruction, while an
osteotomy of the mandibular body was
made in 88 patients (98.9%), who had
reconstruction of segmental defects by
bifocal, trifocal or compression DO. All
patients in this group had a distraction
rate of 1 mm daily. In the TMJ sample,five patients had a distraction rhythm of
twice a day, and one patient once a day.
In 84 (94.4%) of the 89 patients who
underwent segmental bone reconstruc-
tion through bifocal, trifocal bone trans-
port DO or compression DO, distraction
rhythms varying 24 times a day were
used. Data regarding the latency period
were reported in 43 patients (97.7%) whohad bifocal or trifocal bone transport
DO for reconstruction of segmentaldefects; 37 patients (88.3%) had a
latency of 1012 days. Only one patient
in the TMJ sample had information on
the latency period, which was 5 days. All
patients who underwent compression
DO had a latency period of 1012 days.
Data regarding the contention period
were available in 43 patients (97.7%),
who underwent segmental bone recon-
struction through bifocal or trifocal DO;
4 weeks, 1.52.5 months and 3 monthscontention periods were reported in 5
(11.6%), 37 (86.1%) and 1 (2.3%) patient,
respectively. A total of 33 patients
(73.3%) in the compression DO series
h a d a 12 months contention period,
while 12 (26.7%) had one of 1.52.5
months. In the TMJ sample, data were
only available in three patients; they all
had a contention period of 56 weeks. In
the TMJ sample, all patients had exter-
nal devices; five had monodirectional
devices and one had a bidirectional
device. All patients who underwent seg-
mental bone reconstruction throughbifocal and trifocal DO had external
devices, however only nine (20%) had
further information on the type of
device; four patients had monodirec-
tional, three bidirectional devices and
two multidirectional devices. In the com-
pression DO sample, all patients had
external devices but no information
regarding the type of the device wasreported. The amount of lengthening
was mentioned in all patients whounderwent segmental reconstruction by
Table 6. Treatment protocols for mandibular distraction osteogenesis
Type of surgery Rate Latency Contention Device
Mandibular lengthening
Mandibular micrognathia16 y**** Corticotomy 1/d 57 d 68 w E
IOsteotomy 1/d 57 d 68 w E
IMandibular retrognathia
>14 y Body osteotomy 1/d 57 d 68 w IMandibular widening
>12 y Symphyseal osteotomy 0.751/d 57 d 68 w IMandibular alveolar reconstruction>16 y Segmental osteotomy 1/d 57 d 8 w I
0.5/d 57 d 46 m IMandibular bone transportTMJ reconstruction>16 y Reverse-L osteotomy 1/d 57 d 56 w E
ISegmental defect reconstruction>16 y Body osteotomy 1/d 1012 d 68 w E
*DO can be performed in newborns with upper airway obstruction who are tracheostomy-dependent.**Ideal age group to perform DO in patients with severe craniofacial anomalies.***DO should only be performed in severe anomalies who hadnt prior surgery or eventually a secondary distraction in case of growth deficiency
after a costochondral graft86
.****DO can be performed in cases with delayed treatment or residual postsurgical growth disturbance.Distraction implant that will be used for prosthetic treatment after the contention period.Physical therapy during the activation and contention period to induce functional remodeling for the creation of a neocondyl.
94 Swennen et al.
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internal distraction device. One patient
underwent maxillary advancement byDO using the RED system with adistraction rate of more than 1 mm. In
the maxillary alveolar reconstructiongroup, two patients were distracted at
1 mm daily (twice 0.5 mm) using aninternal monodirectional distractiondevice andfive were distracted at 0.5 mm
daily through the use of a distractionimplant. The 45 day latency period was
the most frequent for maxillary advance-ment DO, while all patients who under-
went alveolar reconstruction had oneof 57 days. Of the 122 patients thatunderwent maxillary advancement, data
regarding the contention period werereported in 116 patients (95.1%); 56
patients (48.3%) had a contention periodof 23 months and 60 patients (51.7%) acontention period of 24 weeks. Of those
60 patients, however, maxillary DO wasperformed in 54 using a rigid external
distraction device followed by night-timeretention with a facial mask during
48 weeks, while the other six patientswere distracted using a facial mask fol-
lowed by further orthodontic contentionwith elastics for a period of 3 months. Inthe alveolar reconstruction group, the
five patients who were distracted with adistraction implant, had a contentionperiod of 46 months, while the two
patients who were distracted with an in-ternal monodirectional distraction device,
had a contention period of 23 months.Data regarding the amount of maxillary
advancement were available in 121patients (99.2%) varying from 1 to17 mm; 50 patients (41.3%) had a distrac-
tion distance of more than 6 mm (36 had
a complete and 14 an incomplete Le FortI osteotomy). All patients who underwentalveolar reconstruction had data on the
amount of lengthening varying from 3 to15 mm; the bone-borne distraction de-
vices provided greater lengthening (715 mm) compared to the implant-borne
devices (35 mm). Follow-up data werereported in 109 patients (84.5%) andranged from 3 months to 3 years, with 79
patients (61.2%) followed up for 6 monthsor more. Data on relapse were reported
in 65 patients (50.4%); two had skeletalrelapse. In 87 patients (67.4%) informa-tion regarding complications was given;
2 complications occurred(Table 5).Thus, it appears recommendable to
perform maxillary advancement fromthe age of 5 years after a complete LeFort I osteotomy using an internal or
external device, a 1 mm distraction rate,a 45 days latency period and a 23
months contention period. In the 513years age group, an incomplete Le Fort I
osteotomy can be performed and a facialmask can be used for mild deformities
(Table 8). Recommendations concerningthe parameters for maxillary expansion(Table 8) are based on the scientific
report of M93, while those formaxillary alveolar reconstruction arethe same as for mandibular alveolar
reconstruction(Table 5).
Simultaneous mandibular and maxillarydistraction osteotogenesis
In this group, 24 patients (2.9%) under-
went simultaneous mandibular andmaxillary DO90,95,96. Of the 24 distrac-
tion procedures, 23 (95.8%) were carried
out in HFM patients and one in a TCpatient. Data on age were available forall patients and ranged from 11 to 32
years. All 24 patients had mandibulardistraction procedures with simul-
taneous maxillary DO through IMF.Two different surgical techniques werereported: 21 patients (all HFM) under-
went a unilateral mandibular ramuscorticotomy in combination with a com-
plete Le Fort I osteotomy at the affectedsite and an incomplete Le Fort I oste-
otomy at the unaffected site90,95; threepatients, two HFM and one TC, under-went a unilateral and bilateral osteo-
tomy, respectively, of the mandibularramus combined with a bilateral com-
plete Le Fort I osteotomy96. All patientshad a distraction rate of 1 mm daily anda latency period of 5 days. Contention
periods of 46, 7 and 810 weeks werereported in eight (33.3%), one (4.2%)
and 15 patients (62.5%), respectively.Distraction of the mandibular ramus
was performed in 21 patients (87.5%)using an external monodirectional device
and in three patients (12.5%) usingan internal monodirectional device.Data on mandibular lengthening were
reported in all patients varying from 12to 28 mm, while only 21 patients haddata on the degree of maxillary rotation
varying from 12 to 18 degrees. Follow-up was only reported in nine cases
(37.5%) and ranged from 4 to 25months. Data regarding relapse wereonly mentioned in two patients (8.3%),
showing no skeletal relapse. Informationon complications was reported in
Table 8. Treatment protocols for maxillary distraction osteogenesis
Type of surgery Rate Latency Contention Device
Maxillary advancement
513 y Incomplete Le Fort I 700/900 g 45 d 23 m Facial mask*Complete Le Fort I 700/900 g 45 d 23 m Facial mask*
1/d 45 d 24 w** RED1/d 45 d 23 m I
1316 y Complete Le Fort I 1/d 45 d 24 w** RED1/d 45 d 23 m I
>16 y Complete Le Fort I 1/d 45 d 24 w** RED1/d 45 d 23 m I
Maxillary expansion
>14 y Incomplete Le Fort I*** 0.33/d 57 d 36 m I/bone0.251/d 57 d 36 m I/tooth
Maxillary alveolar reconstruction>16 y Segmental osteotomy 1/d 57 d 2 m I
0.5/d 57 d 46 m I
Complete Le Fort I: pterygomaxillary disjunction is performed/Incomplete Le Fort I: the pterygomaxillary suture and septal base remain intact.*A facial mask should only be used for mild deformities (anterior crossbite
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only eight patients (33.3%); only onecomplication occurred (Table 5).
This procedure can be performedfrom the age of 12 years using a 1 mm
distraction rate, a 5 day latency period,a 2 month contention period and anexternal or internal mandibular device in
combination with IMF. We further sug-gest that a mandibular device can be
combined with an internal maxillarydevice, instead of IMF. Both surgical
procedures can be performed(Table 9).
Midfacial and/or cranial distractionosteogenesis
A total of 96 patients (11.6%) underwentDO procedures involving the midface
and/or cranium5,7,1012,14,15,1721,27,48,49,68,71,77,81,100,125,131,136 (Table 3). Resultsof midfacial and/or cranial DO par-
ameters are shown inTable 10.Data onage were reported in 88 patients and
varied from 3 months to 35 years; 32patients were distracted at 47 yearswhile 28 patients underwent DO of the
midface and/or cranium under the age of4 years; 19 of those were distracted
under the age of 1.5 years. A total of 62patients (64.6%) had midfacial advance-ments, while 25 had monobloc (MB)
advancements after a MB osteotomy.Below the age of 4 years MB DO was the
most performed surgical technique in atotal of 19 patients (12 were below the
age of 1.5 years), while in the other agegroups the Le Fort III procedure wasthe most frequent. Three patients
with Aperts Syndrome underwent com-bined MB and facial bipartition (FB)
Table 9. Treatment protocols for simultaneous mandibular and maxillary distraction osteo-genesis
Type of surgery Rate Latency Contention Device
>12 y Ramus corticotomy 1/d 5 d 2 m E/monoLe Fort I osteotomy* IMF
Ramus osteotomy 1/d 5 d 2 m I/monoLe Fort I osteotomy** IMF
*Complete Le Fort I osteotomy at the affected site, and incompleted Le Fort I osteotomy at the
unaffected site.**Complete Le Fort I osteotomy at both the affected and unaffected site.
Table 10. Midfacial and/or cranial DO: DO parameters related to indication
Crouzons Aperts PfeiffersOtherCST Clefts UniCFM Total %
Age
12 y 4 3 1 1 9 10.288 100
Surgical technique
Monobloc osteotomy 2 8 8 7 25 26.0Fronto-orbital osteotomy 2 3 5 5.2Coronal craniectomy 1 2 3 3.1Bitemporal parietal craniotomy 1 1 1.0
Le Fort III 31 7 4 8 5 2 57 59.0Modified midfacial osteotomy* 2 2 2.1Le Fort II 1 1 1.0No osteotomy 2 2 2.1
96 100Distraction rate
1 mm/d 7 6 5 15 5 1 39 40.6>1 mm/d 27 11 7 1 3 1 50 52.1
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procedures through DO below the age of
4 years18,20. Different distraction rateswere used; 39 patients were distracted at
1 mm daily, 50 patients at more than1 mm (1.53 mm/d) and 7 patients at less
than 1 mm (0.50.8 mm/d). In the agegroup below 4 years of age, 1 mm dailywas the most frequent distraction rate,
while in all other age groups usually adistraction rate of more than 1 mm daily
was used. Data on distraction rhythmwere reported in only 47 cases (49.0%);
five patients had a rhythm of 1 mm once
and nine a rhythm of 0.5 mm twice, while
with a distraction rate of more than 1 mmdaily, activation was performed alwaystwo or three times daily. Two patients
who underwent cranial DO through theuse of implantable springs had a continu-
ous distraction rhythm77. Latency periodswere reported in 82 cases and ranged from07 days. A total of 44 patients had a
latency period of 0 days while 25 patientshad a latency period of 57 days. In the
age group below 4 years, a total of 15patients had a latency period of 57 days,
while in all other age groups no latencyperiod was the most common. Data re-lated to the contention period were men-
tioned in 76 patients and ranged from 3weeks to 6 months; 38 patients had a con-
tention period of 6 months and 28 patientshad one of 23 months. In the age groupbelow 4 years of age, the 23 month con-
tention period was the most frequent,while in all other age groups the 6 month
contention period was the most common.
In three patients (3.1%) immediate osteo-synthesis was made at the end of the acti-vation period. Data on the obtaineddistraction distance were reported in 76
patients (79.2%) and varied from 7.5 to35 mm; 74 patients were distracted more
than 10 mm. A total of 13 patients (13.5%)were distracted through the use of externaldevices; 10 had monodirectional, 3 multi-
directional devices. In 83 patients (86.5%)internal devices were used; 80 had mono-
directional, one a bidirectional device andtwo implantable springs. Length of follow-
up was reported in 58 patients (60.4%) and
ranged from 2 months to 4 years, with only36 cases (37.5%) followed-up for 1 year or
longer. Data on relapse were only reported
in 19 cases (19.8%); 17 had no relapse
and two had mild relapse. Information
regarding complications was mentionedin 87 patients (90.6%); 25 complicationsoccurred (Table 5).
Under the age of 4 years, it appearsthat MB DO is mostly indicated as aresult of functional reasons (respiratory
problems, severe exophthalmos) andcan be performed after a complete MB
osteotomy using internal or externaldevices, a 1 mm distraction rate, a 57latency period and a 23 month conten-
tion period. Midfacial DO is indicatedfrom the age of 4 years in patients who
underwent early conventional fronto-cranial remodeling and advancement,after a complete Le Fort III osteotomy
using the same parameters as the mono-bloc procedure or using the con-
tinuous rapid midface DO parameters(Table 11).
Other types of DO of the craniofacialskeleton
Occasional reports on other types of DOof the craniofacial skeleton include
zygoma distraction21; orbital distrac-tion14, Le Fort II distraction48, nasal
bone distraction in hypertelorism74 anddistraction of scarred soft tissue beforesecondary bone grafting148.
Discussion
Craniofacial DO is a rapidly growing fieldof craniofacial reconstruction that has be-
come a accepted method worldwide forthe treatment of numerous congenital andacquired craniofacial anomalies. Clinical
parameters that affect treatment outcomeof craniofacial DO, include: (1) age; (2)
surgical technique; (3) distraction rate andrhythm; (4) latency period; (5) contentionperiod and (6) distraction device. Distrac-
tion osteogenesis, parameters, however,are not rigidly established and consider-
able variation exists between differentcraniofacial groups. In this article we have
attempted to provide standards for thesevariables and to develop treatment proto-cols for each type of craniofacial DO
based on the review of clinical and ex-perimental investigations reported in the
literature.Craniofacial DO has been reported
successfully in the paediatric, adolescent
and adult patient population. Patient ageat the time of distraction is the most im-portant single variable with potentially
the mostprofound effect on the outcome
of DO51. The human face does not de-
velop as an individual unit but as one
functional unit with growth of one area
depending on growth of the remaining
areas33. C& S8, showed in their
study of mandibular DO in 14 childrenwith a follow-up of 36 years that cranio-facial growth postdistraction was vari-
able; this was also observed by othercraniofacial teams39,51,75. The growth pat-
tern is modulated by both the originalgenetic predispostion of the native boneas well as the accompanying soft tissue
functional matrix39,40. Although the issueof overcorrection remains controversial,
overcorrection seems to be necessary ingrowing patients and younger patients
with greater future growth potential re-quire a greater amountof overcorrectionthan older patients39,40,86.
One of the most critical components ofDO is the surgical separation of thebony
fragments. The original I5456
concept of necessity of preserving endos-teum and periosteum has proved not to be
essential in DO of the human craniofacialskeleton. The literature on craniofacial
DO, however, is sometimes confusingregarding the terms corticotomy/osteo-
tomy, especially concerning mandibularlengthening procedures. Corticotomy isfrequently used, while in reality a com-
plete division of the bone is performed.Therefore, we interpreted the technique of
performing a corticotomy of the buccalcortex with a bicortical notch followed by
a peroperative greenstick fracture as acomplete separation of two bone seg-ments and hence as an osteotomy. In
patients with congenital and acquiredmandibular micrognathia, both an osteo-tomy and a corticotomy have been per-
formed successfully; 189 patients under-went an osteotomy (57.3%) and 141
(42.7%) a corticotomy. We suggest thata complete osteotomy will make the
distraction procedure more reliable,predictable and comfortable for thepatient.
Table 11. Treatment protocols for midfacial and/or cranial distraction osteogenesis
Type of surgery Rate Latency Contention Device
12 y Le Fort III 1/d 57 d 23 m I/E
>1/d** 0/d** 6 m** IMonobloc* 1/d 57 d 23 m I/E
*Monobloc (MB) osteotomy consists of a Le Fort III osteotomy and frontal osteotomy and canbe combined with facial bipartition (FB) in case hypertelorbitism and brachcephaly with facial
flatness.**Continuous rapid midface DO according to Chin and Toth12,136: intraoperative advance-ment up to 10 mm, followed by immediate distraction over 3 to 5 days and a prolongedcontention period.
98 Swennen et al.
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The optimal rate of distraction, ac-
cording to adult orthopedic DO, is 1 mm
daily for each callusfield. Faster distrac-tion rates in enchondral bone result in
local ischaemia in the distraction gapand delayed ossification or pseudarthro-
sis, whereas slower distraction rates re-sult in premature ossification and
consolidation
56
. It has been suggested thatthe membranous bone of the craniofacial
skeleton behaves differently and that there
is a risk for premature consolidation if the
distraction rate is too slow, especially in
very young children where faster distrac-
tion rates comparing to DO of the enchon-
dral bones could be necessary136. This
review showed that a distraction rate of
1 mm daily remains the standard for man-
dibular lengthening, bone transport and
compression DO and for maxillary ad-
vancement and simultaneous mandibular/
maxillary DO procedures. A total of 451
patients who underwent these procedures
had data on complications; only six
patients (1.3%) had premature consoli-
dation. Although a distraction rate of
1 mm daily is effective in mandibular wid-
ening procedures, most patients (68.4%)
had a rate inferior to 1 mm (0.751 mm/d);
four premature consolidation occurred, al-
though not due to the slower distraction
rate but owing to a failure of the distrac-
tion device. Of the 23 patients that under-
went alveolar reconstruction procedures, a
total of 12 (52.2%) had a distraction rate of
1 mm daily, while 11 (47.8%) had a rate of
0.5 mm daily. None of these patients had
premature consolidation. Furthermore,
this review showed that in midfacial and/or
cranial DO, a distraction rate of 1 mm was
the most frequent in patients under the age
of 4, while in all other age groups, faster
distraction rates (1.53 mm/d) were re-
ported. Pseudarthrosis occurred in none of
the 87 patients who had data on compli-
cations. Success of DO not only depends
on the rate of distraction, but also on the
rhythm. I56 showed that osteo-
genic activity was greater when DO was
performed at 0.25 mm four times per day,
as opposed to 1 mm once a day, and even
greater when an autodistractor was usedthat allowed continuous distraction. Based
on our analysis of clinical articles on DO,
no conclusions could be made because of
lack of appropriate data regarding distrac-
tion rhythm. We suggest that several acti-
vations (24) a day, however, are certainly
more comfortable for thepatient.
According to I54,55 success ofDO depends on the response of the initial
callus to tensile stress. Therefore, in theenchondral bone, a latency period of 57days after surgery is necessary to allow
time for initial callus formation and heal-
ing of soft tissues5456. The membranous
bones of the craniofacial skeleton, which
are thin and have a rich blood supply aresignificantly different. Therefore, shorterlatency periods could be acceptable. This
review also showed that in craniofacialDO the 57 day latency period was the
most frequent. Even longer latencyperiods (712 days) were mentioned inpatients who underwent bone transport
and compression DO. On the contrary,shorter latency periods were reported in
maxillary, midfacial and cranial advance-ment DO procedures. A 45 day latency
period was most common reported formaxillary advancement. In the midfacialand cranial DO group a latency period of
57 days was the most reported in the agegroup below 4 years, while in all other age
groups distraction was most frequentlyimmediately started. However, 38 (63.3%)of those patients underwent continuous
rapid midface DO after a Le Fort IIIosteotomy and were all reported by the
same craniofacial team10,12,136. The highfrequency emission of the latency periodis thus not a widely agreed standard, but
results from the high numbers of casescontributed by a single team. We suggest
that a latency period to allow initial callusformation and healing of the soft tissues is
also necessary in DO of the craniofacialmembranous bones.
After the active period of distraction,the distraction appliances are left in placefor adequate consolidation and matur-
ation of the bony callus. This review re-vealed that the 68 week contention pe-
riod was the most appropriate for allmandibular lengthening and expansion
DO procedures and for the reconstructionof segmental defects by bone transport orcompression DO. Longer contention
periods were reported in maxillary (23months), midfacial and/or cranial DO
(23, 6 months). We suggest that despitethe good vascularization of the midfacialcomplex, longer contention periods are
necessary compared to the mandibleowing to the thin structure of the bone at
the distraction sites.Distraction devices used range from
traditional extraoral monodirectional
appliances as introduced byMCet al.83 tobidirectional6466,88 and multi-directional67,68,85,103,104 devices that per-
mit interceptive distraction. Continuedresearch has lead to the development
of intraoral devices, that can betooth26,41,60,61,142, implant37,38,69, orbone11,29,30,41,47,9193,140, supported,
and subcutaneous15,1719 devices used inmidfacial and cranial DO. It is crucial
for the device to be chosen with consid-
eration to the type of skeletal deficiency
and the patients needs. Devices withideal bone-device and bone-tooth move-
ment ratios of 1:1 are recommended.Craniofacial DO was found to be a
procedure with low peroperative andpostoperative morbidity. Data on com-
plications were mentioned in 604(73.0%) of the 828 patients who under-went craniofacial DO procedures.
Although a total of 133 patients (22.0%)had complications, most of these were
mechanical problems due to the distrac-tion appliance or minor local infections(Table 5). Only four hypertrophic scars
due to external devices and one pseudar-throsis were reported. It was remarkable
that only two (8%) cases of meningitiswere reported in monobloc DO proce-
dures. All authors noted that this was incontrast to conventional MBprocedureswhere the morbidity is higher17,18,20,136.
The growing body of literature oncraniofacial DO means that there is a
need for success criteria to provideobjective data in the future. In thisarticle we attempted to establish such
criteria that should be adequate fordeterming long-term functional and
aesthetic results, responding to the needsof our patients (Table 12). The literatureprovides a wide spectrum of useful
tools that can help to increase success,such as stereolithographic models112,130,
3D-cephalometric treatment plan-ning75,135, geometric models78,110,111,
114,129, computer-aided surgery141, ultra-sound35,36,58,80 and endoscopy109,138.The key to success for each type of
craniofacial DO, however, is carefultreatment planning39,40,111. Callus
manipulation can be helpful because itpermits fine-tuning of the occlusionduring distraction44,50,86.
Craniofacial DO has had an enormous
impact and will probably play a major rolein the future in the treatment of congenitaland acquired deformities of the craniofa-
cial skeleton. Through the use of DO,craniofacial deformities can be treated less
invasively. Because there is no need for asecond surgical site to harvest bone, oper-ating time, risk for blood transfusion,
postoperative morbidity and hospital-ization time are reduced. In addition, sim-
ultaneous expansion of the soft tissuefunctional matrix may improve long-term
skeletal stability. The literature dealingwith DO of the craniofacial skeleton, how-ever, shows a lack of long-term data, es-
pecially regarding skeletal relapse. Only479 patients (57.9%) had data on
follow-up and in only 248 patients (30%)
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