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British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739

Systematic review
Transport distraction osteogenesis compared with
autogenous grafts for ramus-condyle unit reconstruction in
temporomandibular joint ankylosis: a systematic review and
meta-analysis
Ashutosh Kumar Singh a,⇑, Anson Jose b, Nikita Khanal c, K.C. Krishna a, Rajib Chaulagain d,
Ajoy Roychoudhury e
a
Department of Oral and Maxillofacial Surgery, TU Dental Teaching Hospital, MMC, Institute of Medicine, Kathmandu, Nepal
b
Oral and Maxillofacial Surgery, Private Practice, New Delhi, India
c
Ek Ek Paila Foundation, Kathmandu, Nepal
d
Department of Oral Biology, Chitwan Medical College, Bharatpur, Nepal
e
Department of Oral and Maxillofacial Surgery, CDER, AIIMS, New Delhi, India

Received 25 August 2021; revised 25 November 2021; accepted in revised form 13 December 2021
Available online 15 December 2021

Abstract

This systematic review was planned to assess the clinical outcomes of transport distraction osteogenesis (TDO) compared with autoge-
nous grafts for reconstruction of the ramus condyle unit (RCU). We searched Medline, Embase, Cochrane Library, Clinicaltrial.gov, and the
references of included trials. The primary outcome was maximal incisal opening (MIO). Of the 148 studies retrieved, five were included
(TDO = 49, autogenous grafts =123). The mean difference in MIO between TDO and autogenous graft RCU reconstruction, based on
the random-effects model was 1.28 mm (95% CI 0.167 to 2.403) in favour of TDO. Re-ankyosis was observed in four cases in the costo-
chondral graft group and none in the TDO group. Reconstruction of the RCU using TDO is comparable to autogenous grafts after the release
of TMJ ankylosis, though the evidence is weak considering the small number of trials, high risk of bias, and absence of long-term results.
Ó 2021 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: TMJ ankylosis; transport distraction osteogenesis; RCU reconstruction; systematic review; meta-analysis; evidence synthesis

Introduction fected.1–7 Regardless of technique, its aims remains the


same, namely, to restore mandibular form, morphology,
Reconstruction of the ramus-condyle unit (RCU) after resec- and masticatory function, reduce disability, and allow
tion of ankylosis has been attempted with multiple strategies, growth in paediatric patients while preventing donor-site
but the ideal method of reconstruction has not yet been per- morbidity and excessive treatment cost.8 The ideal technique
should achieve all of these with the least complications.
There are various reported techniques of RCU reconstruc-
Abbreviations: RCU, Ramus condyle unit; DO, Distraction osteogenesis;
TDO, Transport distraction osteogenesis; TMJA, Temporomandibular joint tion, from autogenous grafts such as costochondral grafts
ankylosis; CCG, Costochondral graft; MIO, Maximal incisal opening. (CCG), sternoclavicular grafts (SCG), free fibula, and allo-
⇑ Corresponding author at: Department of Oral and Maxillofacial plastic materials.9–12 Autogenous grafts are riddled with
Surgery, TU Dental Teaching Hospital, MMC, Institute of Medicine, donor-site morbidity, unpredictable growth, graft fracture,
Kathmandu, Nepal.
E-mail address: dr.ashutosh@iom.edu.np (A. K. Singh).
failure in multiple operated cases, or in scarred tissue, hetero-
topic bone formation, occlusal instability, and a need for the
https://doi.org/10.1016/j.bjoms.2021.12.051 patient to be kept under intermaxillary fixation (IMF).3,7
0266-4356/Ó 2021 The British Association of Oral and Maxillofacial Transport distraction for RCU reconstruction was intro-
Surgeons. Published by Elsevier Ltd. All rights reserved. duced by Stucki-McCormick.13 It later presented good

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732 A.K. Singh et al. / British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739

results in case series by Schwartz and Relle14 and Schwartz15 Study eligibility
and has grown in popularity since then. Kaban et al even
modified their protocol for the management of TMJ ankylo- RCTs, uncontrolled clinical trials, and prospective or retro-
sis in the paediatric age group to include TDO as the choice spective comparative studies published in English were
for reconstruction of the RCU.16 included. Our primary outcome of interest was MIO and
DO is a process of new bone formation between living oss- re-ankylosis after a minimum of six months’ follow up.
eous surfaces that are incrementally and gradually separated Our reported outcomes were also reviewed, including the
according to a predetermined protocol.17,18 In TDO, a bone success of neocondylar formation, laterotrusion and protru-
segment is created and slowly moved away from the host bone sion, deviation of function, facial symmetry, and
towards a docking bone surface at the other side of the defect. complications.
In transport disc DO, new bone forms at the trailing edge of the
transport disc, while a fibrocartilage cap forms at the leading Assessment of risk of bias
edge.19,20 In reconstruction of the RCU, after resection of the
ankylotic mass, a transport disc is created from the remaining A generic and modified version of the Cochrane Collabora-
ramus stump by an L osteotomy and distracted towards the tion’s tool for assessing the risk of bias (ROB 2 tool) for com-
glenoid fossa. The regenerate towards the ramus stump forms parative studies was used to assess the risk of bias at the
the RCU, while the fibrocartilage cap towards the glenoid study level. Two reviewers (AKS and RC) assessed the risk
fossa becomes an articular surface.21,22 Multiple cohort stud- independently, and any inconsistency was resolved by
ies have reported the advantageous outcomes of TDO, but few discussion.
comparative studies and clinical trials have reported on out-
comes compared with other techniques of RCU reconstruc- Statistical methods for meta-analysis
tion.23–28 Despite being an option for this, TDO has not
become as popular as expected and, to our knowledge, conclu- The analysis was carried out using the mean difference of the
sive evidence of its efficacy compared with that of other primary outcome using a random-effects model. The amount
modalities of RCU reconstruction are few.23,24 of heterogeneity (tau2), was estimated using the DerSimo-
nian and Laird estimator. Studentised residuals and Cook’s
Method distances were used to examine outliers and/or influential
studies in the context of the model. Studies with a Cook’s
Design and search strategy distance larger than the median plus six times the interquar-
tile range of the Cook’s distance were considered to be influ-
This systematic review protocol was registered in PROS- ential. The rank correlation test and the regression test, using
PERO (CRD42021258236). The PICOS strategy for hypoth- the standard error of the observed outcomes as the predictor,
esis generation and search strategy was: were used to check for funnel plot asymmetry. All the anal-
yses were carried out using the metaphor package, Meta-
 (P) Patients with TMJ ankylosis planned for RCU reconstruc- Analyisis Package for R.29
tion after resection of the ankylosis
 (I) Intervention: TDO for RCU reconstruction Results
 (C) Comparator or control: other modalities of RCU
reconstruction
We identified 148 articles that were verified for eligibility.
 (O) Primary outcomes: maximal incisal opening (MIO), recur-
Five comparative clinical studies met our inclusion crite-
rence of ankylosis. Secondary outcomes: other reported out-
comes and complications ria.3,4,23,24,30 The PRISMA flow diagram for selection of
 (S) Study design: studies in humans, including randomised con- the studies is presented in Figure 1.
trolled trials (RCTs), uncontrolled clinical studies, and retro-
spective or prospective comparative studies Study characteristics

We searched Medline (OVID), Embase (OVID), the One study compared TDO, CCG, and Surgibone (Unilab) as
Cochrane Library, Clinicaltrials.gov, Google Scholar, and treatment modalities to reconstruct the ramus joint complex
grey literature up to 31 July 2021. The search strategy used in patients with ankylosis of the temporomandibular joint
for the Medline search is presented in Appendix A. Two (TMJ),4 three studies compared TDO with CCG,3,23,30 and
independent reviewers (AKS and AJ) performed the search, one compared TDO with SCG.24 Three mentioned the fund-
and any inconsistency was resolved by discussion. We ing source, three declared that they had been given no fund-
extracted data on the author, country, region, type of study, ing for the study, and six did not provide any information on
baseline characteristics, type of ankylosis, methodological funding. The potential conflict of interest of the investigators
rigour of randomisation and blinding of outcome assessors, was not mentioned in two. Study characteristics are detailed
technique of transport distraction, and primary as well as sec- in Table 1
ondary outcomes.

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A.K. Singh et al. / British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739 733

Fig. 1. PRISMA flow diagram of trial selection.

Risk of bias assessment in the CCG group compared with 37.12 in the TDO group. In
the study by Elgazzar et al,4 postoperative MIO was 34.8 mm
The overall risk of bias was high in the included studies. for TDO, 33.1 mm for CCG, 34.6 mm for CCG with BPF,
Only the study by Kaur et al23 fulfilled the criteria for a and 34.5 mm for coronoid process graft. Figure 4 shows a
low-bias study; the others failed to mask the outcome asses- forest plot of the outcome of MIO.
sors, which can introduce reporting bias. The risk of bias in
the studies is presented in Figure 2, and an overall risk of bias Re-ankylosis and occlusion
across the studies in Figure 3. The risk of bias analysis is also Kaur et al found no re-ankylosis or open bite in the CCG or
presented in Appendix B. TDO group.23 Elgazzar et al4 reported that two recurrent
cases (2%) in the CCG group were successfully managed
Qualitative analysis of outcomes with DO. There were no cases of re-ankylosis in the TDO
group. Jiang et al30 reported no cases of re-ankylosis in both
Maximal incisal opening (MIO) the CCG and TDO groups, whereas Sahoo et al reported two
In the study by Kaur et al,23 the mean (SD) MIO increased cases in the CCG group and none in the TDO group.3
from 8.5 (4.1) mm to 35.7 (2.7) mm with CCG and from
9.5 (7.1) mm to 34.4 (8.9) mm with TDO. This was statisti- Lateral and protrusive mandibular movements
cally significant (p<0.005) but differences between the In the study by Kohli et al,24 the intergroup difference
groups were not. In the study by Kohli et al,24 there was between SCG and TDO was not statistically significant
no significant intergroup difference in mean MIO. Jiang (p>0.05). Kaur et al23 reported a significant improvement
et al30 reported MIO of 29.5 (2.7) in the CCG + CPG (coro- from a preoperative median of 5.4 and 3.25 mm to 8.3 and
noid process graft) group compared with MIO of 32.1 (2.79) 7.3 mm during ipsilateral laterotrusion in the CCG and
in the TDO group. Sahoo et al3 reported MIO of 36.08 (6.7) TDO groups, respectively, during the follow-up period.

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Table 1

734
Study characteristics.
First author, Country of Study design No. Demographic Type of ankylosis Outcomes Follow up
year, and study character
reference age, gender
Kaur 202023 India RCT TDO=12 Mean (SD) age U/L bony Primary: Median (range) 31.5
CCG=10 10.32 (2.85) MIO (24 – 39) months
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(range 3-16)
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years Secondary: Attrition: 0


occlusion, laterotrusion, protrusion, re-
Gender: ankylosis, neocondyle, chin deviation, facial

A.K. Singh et al. / British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739
Male: 13 asymmetry, midline shift,
Female: 9 neodisc formation.
Kohli 201724 India Prospective TDO=11 Age: 18–48 years U/L bony MIO At 1 week,
comparative SCG=11 Mean- 24.5 years Duration of the procedure and 1, 3 and 6 months
study reconstruction
Gender: Attrition: 0
Male: Female Maximum protrusive and laterotrusive
Ration 1.4:1 excursions

Mean condylar height, width, amount, and


degree of resorption

Pain on function
Elgazzar 20104 Canada Retrospective CCG: 54 Mean (range) age U/L and B/L bony, MIO Weekly for 6 weeks, at 3 months and 6
Egypt study TDO: 11 19.43 (2–41) fibrous and fibro-osseous months and then annually ‘til the end up
Saudi years TMJ ankylosis follow up period.
Arabia Mean: 28.9 months
Gender
Male: 38
Female: 63
Sahoo 20123 India Retrospective TDO = 8 Mean (range) U/L and B/L bony TMJ MIO 1 week, 1 month, quarterly for one year
cohort study CCG = 37 age14.3 ankylosis followed by yearly visits till they lost
(0.5-45.5) years Failure of treatment in terms of re-ankylosis contact

Gender: Overgrowth of costochondral graft,


Male: 38 donor-site morbidity,
Female: 26
Other complications
Jiang 201830 China Retrospective CCG + Mean (range) age U/L and B/L bony MIO 1, 3, 6, and 12 months
cohort study CPG= 11 9.3 (5 – 13) years ankylosis
TDO= 7 Re-ankylosis Mean (range) 24.8 (12 – 50) months
Gender
Male: 7
Female: 11
MIO: maximal incisal opening; CCG: costochondral graft; CPG: coronoid process graft; TDO: transport distraction osteogenesis; MDA: Matthews device arthroplasty
A.K. Singh et al. / British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739 735

et al reported pain scores on a VAS scale of 1-10.24 On the


first postoperative day, 90.9% (10/11) of SCG patients had
a pain score of more than five, while in the TDO group this
was 72.7% (8/11). At the six-month follow up, 72.7% of
SCG patients and 90.9% of TDO patients were pain-free.
Sahoo et al reported an incidence of pleural tear during
CCG harvest.3

Quantitative analysis and publication bias


The reported outcomes are presented in Table 2.
Five studies were included in the meta-analysis. The
observed mean differences ranged from -1.30 to 2.60 mm,
with the majority of estimates being positive (80%). The esti-
Fig. 2. Risk of bias in the included studies. mated average mean difference based on the random-effects
model was 1.28 mm (95% CI: 0.16 to 2.40) in favour of
There was an improvement in contralateral laterotrusion TDO, so the mean difference in MIO between the two groups
from a median of 0.4 mm and 1.58 mm to 2.0 mm and was statistically significant (t(4) = 3.19, p = 0.03) (Fig. 4).
4.33 mm in the CCG and TDO groups, respectively. Protru- According to the Q-test, there was no significant heterogene-
sive movement improved from 0.3 to 1.1 mm in the CCG ity (Q (4) = 2.86, p = 0.58, tau2 = 0, I2 = 0%). Examination
group and from 0.5 to 1.17 mm in the TDO group. of the studentised residuals revealed that none of the studies
had a value larger than ±2.57, hence in the context of this
Complications model there was no indication of outliers. According to
Kaur et al reported pin tract infection in four patients and Cook’s distances, none of the studies could be considered
compression osteogenesis in one in the TDO group.23 Kohli to be overly influential. Neither the rank correlation nor the

Fig. 3. Risk of bias across the included studies.

Fig. 4. Forest plot of the outcome of maximal incisal opening (MIO).

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736
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Table 2
Table of reported outcomes.
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First author, year, Occlusion Mean (SD) maximal Protrusion Laterotrusion Distraction Complications Neocondyle Ramus
and reference incisal opening (mm) (mm) achieved (mm) height

A.K. Singh et al. / British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739
Kaur 202023TDO Pin tract infection TDO group-4
No open bite 34.4 (8.9) 1.17 7.3 mm ipsilateral/ Mean (SD) Compression osteogenesis -1 Mean (SD)
CCG No open bite 4.43 mm 10.42 (3.72) Neocondyle fused with the temporal bone and non- 6 43.1 (6.5)
35.7 (2.7) 1.1 contralateral union appeared at junction of the neocondyle to ramus
8.3 mm ipsilateral/ stump-1 45.4 (6.3)
2.0 mm contralateral
Kohli Anterior open bite in 16 patients
201724SCG Open 31.8 2.6 5.0 mm ipsilateral/ 24.7 Post distraction imbalance in 3 patients in TDO group Not
TDO bite=16 32.1 2.3 1.9 mm contralateral reported
4.1 mm ipsilateral / 8
1.4 mm contralateral 3
Sahoo 20123 Not reported Not reported Not reported Not reported Pleural tear -1 Not reported Not
CCG Re-ankylosis reported
TDO 36.08 (6.70)
37.12 (1.95) CCG=2
TDO=0

Jiang 201830 Not reported Not reported Not reported Not reported None Not reported Not
CCG+CPG reported
TDO 29.5 (2.46)
32.1 (2.79)
Elgazzar Not reported Not reported Not reported Not reported Re-ankylosis Not reported Not
20104CCG =54 33.2 (2.42) TDO=0 reported
TDO =11 34.7 (1.9) CCG=2
CCG: costochondral graft; TDO transport distraction osteogenesis; CPG: coronoid process graft
A.K. Singh et al. / British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739 737

regression test indicated any funnel plot asymmetry (p = 0.81 the transport disc. Interestingly, after initial healing, the
and p = 0.44, respectively). cephalic end of the leading edge undergoes resorption and
remodelling to form a near anatomical articular surface cov-
Discussion ered with fibrous tissue. However, in a completely remod-
elled transport disc, the cartilage layer was not sufficient to
Several published studies have analysed the role of TDO in induce growth. Hikiji et al38 reported that the cartilaginous
reconstruction of the RCU.15,25–27,31 A recent narrative sys- cells, which were activated by mesenchymal stimulation as
tematic review elucidated the various applications of DO in a result of surgical trauma, later underwent ossification and
TMJ ankylosis and found a relapse in posterior ramus height subsequent remodelling on the upper surface of the transport
after TDO for ramus reconstruction; it also recommended a disc. Hence it is clear that the transport disc lacks an inherent
meta-anylsis to evaluate TDO in TMJ ankylosis. Hence this growth potential, unlike the CCG.
meta-anaylsis was planned to bring forth comparative results Although the TDO lacks growth potential, the clinical
of TDO in TMJ ankylosis.32 outcomes of DO are comparable with those of reconstruction
The goals of TMJ reconstruction include near anatomical with a CCG.25 Even though the mean maximal mouth open-
restoration of the joint, restoration of ramus height, mainte- ing was constant throughout the study period, the mean abso-
nance of occlusion, and prevention of anterior open bite. A lute ramus height of the reconstructed RCU decreased
variety of treatment modalities have been suggested for significantly in the included studies.24,27 These results sug-
reconstruction of the TMJ, namely CCG, SCG, transport disc gest that the height of the reconstructed condyle was not
DO, iliac bone, vascularised fibula, and alloplastic total joint stable in the long term and resulted in progressive mandibu-
replacement.3,33 The common disadvantages of autogenous lar asymmetry. This could be explained on the basis of
reconstruction of the TMJ include unpredictable growth pat- delayed resorption of the distracted segment. A smaller
terns, donor-site morbidity, resorption, requirement of degree of overall resorption of the transport disc is inevitable,
screws and plates for fixation, and a period of immobilisation and the percentage of resorption increases when the total
for integration of the graft.34 length of vertical distraction is greater than the length of
At present the CCG is the gold standard in TMJ recon- the transport disc. It is therefore advisable to keep the size
struction in paediatric patients.23 The advantages are its rel- of the transport disc to at least 1.5-2 cm to prevent long-
ative rigidity, morphological similarity to the condyle, and term complications.
presence of the hyaline cartilage cap. Above all, its inherent Occlusal derangement and anterior open bite were noted
growth potential makes it suitable for reconstruction of the with TDO in various studies. Xiao et al27 reported five
condyle in growing patients with TMJ ankylosis, as it under- patients (23.8%, 5/21) with anterior open bite during follow
goes adaptive remodelling to form a functional condyle. up. Bansal et al26 reported that 16 of 22 patients (72.7%) pre-
However, its disadvantages in children are the necessity for sented an anterior open bite postoperatively. Anterior open
a second operative site, pneumothorax, graft resorption, an bite is commonly associated with bilateral joint surgeries,
unpredictable growth pattern, and compromised graft healing especially when coronoidectomy is also performed. One fun-
due to scar tissue from previous joint surgery.23,35 damental biomechanical fact that needs to be remembered is
The advantages of DO are manifold. It shares the benefits that the mandible acts as a class III lever and cannot pivot
of autogenous bone grafting without the requirement of a around any point that is anterior to the masticatory muscles.
donor site. Similarly, as with any other reconstruction modal- After the arthroplasty the class III lever is converted into a
ity, a period of immobilisation is not required, which is crit- class I lever with the masticatory muscles acting as a ful-
ical when reconstruction is performed in patients with TMJ crum. Therefore if the coronoid is intact, the majority of
ankylosis and early mobilisation of the jaw is encouraged. patients can still maintain a functional occlusion by selective
Additionally, healing of the TDO is independent of tissue activation of the bilateral temporalis muscle in an oblique
at the recipient site unlike that of other autogenous non- vector that is exactly opposite to the action of the suprahyoid.
vascularised grafts where the scarred surgical site undergoes In the absence of the coronoid /or stripping of the temporalis,
fibrosis and inhibits vascularisation of the graft, which even- particularly in cases of long-standing ankylosis, activity of
tually undergoes resorption. Gradual distraction of the trans- the short suprahyoid musculature exceeds the vertical vector
port segment not only results in elongation of the skeletal provided by the pterygoid-masseteric sling, and results in
tissue, but also helps with the distraction histiogenesis of anterior open bite. This postoperative open bite can be cor-
adjacent tissues.15,23 This results in a more harmonious rected using guiding elastics and/or manual training to bring
lengthening of skeletal, muscular, and soft tissue periosteal about neuromuscular adaptation during the treatment period.
complex, thereby aiding better adaptation of the neocondyle In the present study, only Kaur et al23 evaluated the presence
to the surroundings. of pseudo disc formation after TDO. This autogenerated
Recently, animal studies have demonstrated the ability of pseudo disc prevents interference of the transport disc with
TDO to induce biomechanical stimulation leading to the for- the skull base during functional movement of the mandible,
mation of fibrocartilage at the leading edge of the transport and also prevents relapse.
disc.36,37 This cartilaginous matrix is perhaps derived from Some limitations of this systematic review need to be
mesenchymal stimulation of the periosteal tissue overlying addressed. First, we found only a limited number of compar-

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738 A.K. Singh et al. / British Journal of Oral and Maxillofacial Surgery 60 (2022) 731–739

ative studies in the literature. Secondly, there was a lot of 5. Zhi K, Ren W, Zhou H, et al. Management of temporomandibular joint
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Appendix A. Supplementary data combined with orthodontic treatment in a patient with unilateral
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