Int. J. Oral Maxillofac. Surg. 2010; 39: 573579 doi:10.1016/j.ijom.2010.03.021, available online at http://www.sciencedirect.

com

Clinical Paper Rconstructive Surgery

Two-step transport-disk distraction osteogenesis in reconstruction of mandibular defect involving body and ramus
J. Chen, Y. Liu, F. Ping, S. Zhao, X. Xu, F. Yan: Two-step transport-disk distraction osteogenesis in reconstruction of mandibular defect involving body and ramus. Int. J. Oral Maxillofac. Surg. 2010; 39: 573579. # 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. One-step transport-disk distraction osteogenesis (TDDO) is effective for repairing segmental mandibular defects. The authors studied whether it was effective for reconstructing angled large mandibular defects using a two-step TDDO procedure in seven patients suffering from neoplasm. In the two-step TDDO procedure, the rst distraction (horizontal distraction) was initiated immediately after mandibulectomy, aimed at restoring the mandibular body. It was followed by the second distraction, which was obliquely vertical and aimed at restoring the height of the ramus. The distraction rate was set at twice 0.4 mm/day. The treatment lasted for 1418 months. The horizontal distraction length ranged from 48 to 55 mm, and the vertical one from 33 to 43 mm, with full ossication in the distraction area. No obvious shift of mandible, malocclusion or mouth opening limitation was observed. Patients had a regular diet and spoke clearly. In conclusion, the two-step TDDO is still an option for the reconstruction of large angled mandibular defects when patients are prudently selected, despite the long treatment period required.

J. Chen1, Y. Liu1, F. Ping1, S. Zhao2, X. Xu1, F. Yan1

1 Department of Oral and Maxillofacial Surgery, Second Afliated Hospital, School of Medicine, Zhejiang University, Hangzhou, P .R.China; 2Department of Oral and Maxillofacial Surgery, Afliated Hospital of Stomatology, School of Medicine, Zhejiang University, Hangzhou, P .R.China

Keywords: reconstruction; mandible; distraction osteogenesis; transport disk. Accepted for publication 23 March 2010 Available online 28 April 2010

Large mandibular defects resulting from tumor resection lead to severe aesthetic and functional sequelae. The patients postoperative quality of life largely depends on the quality of the mandibular reconstruction. Vascularized or non-vascularized bone grafting has been the mainstay of mandibular reconstruction treatment because of its predictable effect,7,12,15,16 but its use of autogenous bone and the donor site morbidities caused
0901-5027/060573 + 07 $36.00/0

by its harvesting encouraged the authors to seek an alternative1,10. Distraction osteogenesis is often used to correct craniomaxillofacial bone malformations20,21. Transport-disk distraction osteogenesis (TDDO) has been applied clinically to reconstruct craniomaxillofacial bone defects resulting from tumor resection9. In this method, a bone segment is osteotomized adjacent to the defect and then distracted slowly across the defect so

that new bone forms in the continuously widening gap, eventually leading to reconstruction of the bone defect. The distracted bone segment is referred to as the transport disk18. Constantino et al. carried out a series of studies in dogs on the biomechanical properties of reconstruction of mandibular defect with TDDO and concluded that the bone regenerated by TDDO is strong enough to resist the forces of mastication3,4. Since then, clinical cases of

# 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

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mandublar reconstruction with TDDO have been reported5,13,14,22. Despite its success in reparing segmental bone defects, it is impossible for a single TDDO procedure to reconstruct a mandibular defect that involves the body, angle and the whole ramus. There are two difculties: the defect size is beyond the maximal limitation of most internal distraction systems; and the angled shape can not be formed with a single TDDO. In the present study, this type of mandibular defect was reconstructed successfully with a double-step TDDO procedure, in which the rst TDDO was horizontal distraction and aimed at restoring the mandibular body, and the second was vertical distraction and aimed at reconstructing the height of the ramus. The indications, limitations and noteworthy aspects of this method are described.
Patients and methods

Fig. 1. Design of the mandibulectomy and the transport disk on a solid model of the preoperative mandible.

Patients with a unilateral mandibular defect involving the posterior part of the body, the angle and the whole ramus were candidates for this study. They were informed about the options for mandibular reconstruction, including traditional vascularized or non-vascularized bone grafting and the double-step TDDO. Those preferring the double-step TDDO method were involved in the present study. The double-step TDDO method was applied in 7 consecutive patients suffering from benign neoplasm of the mandible. The patients clinical data are given in Table 1.
Ablative surgery and horizontal distraction procedure

All patients underwent CT scanning preoperatively. The scanning data were used to manufacture a solid model using rapid prototyping techniques. Mandibulectomy was simulated and the horizontal distrac-

tion procedure (including size of the transport disk, the distraction orientation and length; Figs. 1 and 2) was planned with the solid model. A sub- and retro-mandibular incision was made combined with an intra-oral incision under general anesthesia. The soft tissues around the lesional mandible were elevated. The elevation was performed in the subperiosteal plane in an area where the periosteum was beyond a safe surgical margin. Otherwise, the periosteum had to be sacriced. Extensive elevation of the healthy mandible should be avoided. Especially for the bone that was to be made into the transport disk, elevation of the tissues on the medial side of the healthy mandible had to be strictly prevented. Mandibulectomy was performed beyond the safe surgical margin (0.5 cm from the lesion) and the lesional mandibular block was removed, including the whole ramus, the angle and the posterior part of the body (Fig. 3).

The intra-oral incision was sutured in two layers; the oral mucosa and the submucosal layer. A transport disk of about 14x18 mm was osteotomized on the mandibular stump. Special attention was paid to avoid lacerating the soft tissues attaching to the medial side of the transport disk. The internal distraction device (Cibei Med, Cixi, China) was xed to the transport disk and the remaining mandible (remaining basal bone), with the transport segment in close contact with the basal bone (Fig. 4). The activation arm was submandibularly placed and the extra-oral incision was sutured in layers. The distraction was started at the rate of twice 0.4 mm per day after a latency period of 7 days and ended when the expected distraction length was reached. The consolidation period was continued for at least 16 weeks, decided by the degree of calcication in the distraction gap on the panoramic radiograph.

Table 1. Reconstruction of mandibular defect of body and ramus with two-step TDDO in 7 patients. Sex/ Age (ys) F/18 M/22 F/20 M/25 F/19 F/33 F/36
*

Case 1 2 3 4 5 6 7

Diagnosis Ossifying broma Ameloblastoma Ossifying broma Odontogenic keratocyst Ameloblastoma Ameloblastoma Ameloblastoma

Distraction length (mm) horizontal 55 51 48 53 50 47 43 vertical 37 41 40 42 39 36 35

Bone quality Excellent Excellent Excellent Excellent Excellent Excellent Good

Max. insical opening (mm) 40 42 35 35 35 37 31

Midline shift No No No No No No No

Occlusion Good Good Good Mild anterior open bite Good Mild anterior open bite Good

Treatment duration (month) 18 17 16 16 15 14 14

Follow-up (month)* 27 21 13 11 7 5 2

since removal of distraction device for the vertical distraction.

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Vertical distraction procedure

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Fig. 2. Design of the distraction orientation and length of the horizontal distraction by simulative distraction on the solid model.

The vertical distraction procedure aimed to restore the height of the ramus. It was applied at the end of consolidation period of the horizontal distraction procedure. The vertical distraction was also planned on a solid model preoperatively (Fig. 5). Under general anesthesia, the extra-oral incision was reopened to expose the distracted mandibular body and the horizontal distraction device (Fig. 6). Elevation of the soft tissues on the medial side was avoided. After removal of the horizontal distraction device, a transport disk of approximately 1512 mm was cut. The vertical distraction device was then placed and xed (Fig. 7), with the distraction orientation set pointing to the glenoid fossa. The activation arm was transcutaneously placed. The vertical distraction was initiated after a 7 day latency period and continued at a rate of twice 0.4 mm per day until the planned ramus height was reached. A consolidation period then followed. The distraction device was removed after ossication of the distraction gap.

Regular follow-up

Fig. 3. Intra-operative view: mandibulectomy with a Gigli saw.

Patients were followed-up periodically. The follow-up included panoramic radiography, assessment of mastication and facial symmetry, mouth opening, occlusion, speech, and recurrence of tumor. The function of mastication was judged by the diet type (soft or regular diet). Facial symmetry was categorized as symmetrical or unsymmetrical, judged by the doctors observation. It was also reected by the mandibular midline (shift or no shift). Mouth opening was judged by maximal incisal opening. Anterior or lateral open bite was judged to evaluate occlusion. Speech was classied as clear or unclear by the patients pronunciation.
Results

Fig. 4. Placement of the distraction device for the horizontal distraction procedure.

Two-step distraction TDDO treatment was completed in all patients. The distraction length ranged from 43 to 55 mm horizontally and from 35 to 42 mm vertically (Table 1). The treatment period lasted for 1418 months, longer than expected. It was mainly caused by the patients failure to return for visits in time. A consolidation period of 16 weeks was enough, judged by the high-degree ossication on the radiograph (Fig. 8) and the intra-operative view of the new bone (Fig. 9). In the whole treatment period, no distraction device failure or intra-oral

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exposure was found. The incision healed uneventfully, without mucosal dehiscence. Pain and obvious discomfort occurred at the end of distraction in two patients, although it was tolerable. Obvious scars were left on the skin where the activation arm pierced through, which were trimmed later during removal of the distraction device. The patients have been followed up for 227 months (Table 1). In all 7 patients, neither tumor recurrence nor mandibular fracture was observed. Limitation of mouth opening was observed in two patients at the end of the treatment, with maximum incisal opening of 21 and 23 mm. All patients demonstrated satisfactory mouth opening through exercise, with maximum incisal opening from 31 to 42 mm (Table 1). The postoperative appearance of all patients was roughly symmetrical. Obvious lateral shift of the mandible was not found (Fig. 10). The remaining teeth maintained the preoperative occlusion, except in two patients who demonstrated slight anterior open bite and needed maxillomandibular elastic reduction to improve the occlusion (Table 1). The patients could pronounce clearly after a period of exercise. Three patients had received removable partial denture rehabilitation. All patients had a regular diet with the remaining teeth and the partial denture.
Discussion

Fig. 5. Design of the vertical distraction on the solid model. The distraction orientation was set pointing to the glenoid fossa and the distraction length was lled with wax.

Fig. 6. Intra-operative view: new bone generated by the horizontal distraction was fully ossied when the distraction device from the rst step was removed.

Fig. 7. Intra-operative view: placement of the distraction device for the vertical distraction procedure.

Owing to the studies by Constantino et al.3,4, TDDO is now an option for reconstruction of segmental mandibular defects. It is difcult for a single-step TDDO to reconstruct a mandibular defect involving body, angle and the whole ramus, which is large and angled. For this type of mandibular defect, the large size is a problem, while the angled shape is a bigger challenge for restoration. The authors attempted to resolve these difculties using the double-step TDDO in the present study. The posterior part of the body and angle, which was roughly horizontal, was reconstructed through the rst distraction. The second distraction was applied to restore the obliquely vertical ramus. The results show that the shape and functions of the mandible reconstructed with this technique were satisfactory (Fig. 11). It is crucial for the transport disk to acquire enough nutrition and oxygen to maintain vitality, which is a prerequisite for the success of TDDO8,9. In this doublestep TDDO, the transport disk could

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Fig. 8. Panoramic radiography: the distraction gaps of both the horizontal and the vertical distraction were lled with highly ossied new bone at the end of treatment.

Fig. 9. Intra-operative view: the whole distraction gap was lled with high-quality new bone when the vertical distraction device was removed.

Fig. 10. Intra-oral view: occlusion was good and the midline of the mandible did not shift at the end of treatment.

obtain nutrition and oxygen only from the surrounding tissues. Therefore, elevation of the soft tissues on the medial side of the transport disk should be prevented. Periosteum beyond a safe surgical margin should be preserved during mandibulect-

omy to maintain a rich blood supply and osteogenicity of the defect area, a condition conducive to bone regeneration18. Radiotherapy is generally considered to be an adverse factor for bone regeneration, causing damage to the vasculature and

cells, therefore it is not recommended to apply this two-step TDDO procedure to patients who have undergone radiotherapy until more data is obtained from animal experiments. An internal distraction system was exclusively applied in this study, because internal distraction systems produce fewer scars and are more acceptable to patients than external distraction systems11. The activation arm was placed submandibularly rather than intra-orally. Although probably leading to skin scars, it made the daily distraction much easier to manipulate and reduced infection or dehiscence of oral mucosa9. The submandibular scars could be trimmed when the distraction device was removed. Three aspects have to be considered when planning and performing vertical distraction. First, is the size of the transport disk. The bone from which the transport disk is created is generally limited in height, so a balance has to be struck between the size of the transport disk and the height of the remaining basal bone, so that both the two parts are vital and strong enough to resist the force produced by distraction. Second, the distraction orientation has to be set pointing to the glenoid fossa, to restore the ramus-condyle unit, which is important for normal occlusion6,19. Third, the distraction length has to be determined. In the authors experience, a gap of approximately 10 mm between the glenoid fossa and the transport disk should be maintained when treatment is accomplished, working as gap arthroplasty to reduce ankylosis2,17. It seldom led to apparent ramus height insufciency or shift of the mandible, as was demonstrated in this study. Gonzalez-Garcia et al. also applied twostep TDDO to mandibular reconstruction8. Their cases were different from the present ones. Their patients had the condyle reserved, which made it easy to set the vertical distraction orientation. The distraction orientation of the second step was adjusted 20 degree above the rst one in their study, with no intention to restore the angled shape symmetrical to the contralateral side. In contrast, the symmetry of the bilateral mandibular angle was intentionally restored in this study, with the assistance of preoperative simulation on the solid models. This should improve facial symmetry. Autogenous vascularized or non-vascularized bone grafting remains the mainstay for mandibular reconstruction because it ensures satisfactory mandibular function and prole after reconstruction, especially when combined with prefabricated recon-

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3. Costantino PD, Friedman CD, Shindo ML, Houston G, Sisson Sr GA. Experimental mandibular regrowth by distraction osteogenesis. Long-term results. Arch Otolaryngol Head Neck Surg 1993: 119: 511516. 4. Costantino PD, Shybut G, Friedman CD, Pelzer HJ, Masini M, Shindo ML, Sisson Ga Sr. Segmental mandibular regeneration by distraction osteogenesis. An experimental study. Arch Otolaryngol Head Neck Surg 1990: 116: 535545. 5. Elsalanty ME, Taher TN, Zakhary IE, Al-Shahaat OA, Refai M, El-Mekkawi HA. Reconstruction of large mandibular bone and soft-tissue defect using bone transport distraction osteogenesis. J Craniofac Surg 2007: 18: 13971402. 6. Gabbay JS, Heller JB, Song YY, Wasson KL, Harrington H, Bradley JP. Temporomandibular joint bony ankylosis: comparison of treatment with transport distraction osteogenesis or the matthews device arthroplasty. J Craniofac Surg 2006: 17: 516522. 7. Gaggl A, Burger H, Muller E, Chiari FM. A combined anterolateral thigh ap and vascularized iliac crest ap in the reconstruction of extended composite defects of the anterior mandible. Int J Oral Maxillofac Surg 2007: 36: 849853. 8. Gonzalez-Garca R, Naval-Gas L, Rubio-Bueno P, Rodrguez-Campo F.J.. Usandizaga JL. Double-step transport osteogenesis in the reconstruction of mandibular segmental defects: a new surgical technique. Plast Reconstr Surg 2006: 118: 16081612. 9. Gonzalez-Garcia R, Rubio-Bueno P, Naval-Gas L, Rodrguez-Campo FJ, Escorial-Hernandez V, Martos PL, Munoz-Guerra MF, Sastre Perez J, Gil-Diez Usandizaga JL, Diaz-Gonzalez FJ. Internal distraction osteogenesis in mandibular reconstruction: clinical experience in 10 cases. Plast Reconstr Surg 2008: 121: 563577. 10. Hartman EH, Spauwen PH, Jansen JA. Donor-site complications in vascularized bone ap surgery. J Invest Surg 2002: 15: 185197. 11. Hibi H, Ueda M. New internal transport distraction device for reconstructing segmental defects of the mandible. Br J Oral Maxillofac Surg 2006: 44: 382385. 12. Holzle F, Kesting MR, Holzle G, Watola A, Loeffelbein DJ, Ervens J, Wolff KD. Clinical outcome and patient satisfaction after mandibular reconstruction with free bula aps. Int J Oral Maxillofac Surg 2007: 36: 802 806. 13. Kessler P, Schultze-Mosgau S, Neukam FW, Wiltfang J. Lengthening of the reconstructed mandible using extraoral distraction devices: report of ve cases. Plast Reconstr Surg 2003: 111: 14001406.

Fig. 11. 3D CT scanning: the reconstructed left mandible was roughly symmetrical to the contralateral side at the end of treatment.

struction plates. Vascularized bone grafting can also be used to reconstruct the mandible in patients who have undergone radiotherapy. The morbidities caused by harvesting bone aps, which can be severe at the initial stage, has been reduced to a low level following improvements in instruments, the development of microvascular anatomy, and modication of the bone harvesting techniques. In contrast, it is difcult for TDDO to ensure satisfactory postoperative mandibular shape and occlusion. The TDDO technique generally requires a long period and several operations to accomplish reconstruction. This weakness is important when multi-stage TDDO is applied to reconstruct a large mandibular defect. As was demonstrated in this study, the treatment period ranged from 14 to 18 months. The long treatment period hinders its wide application. In spite of its weaknesses, the two-step TDDO technique avoids sacrice of autogenous bone and the donor site morbidities caused by bone (ap) harvesting. This is important for adolescents in whom bone harvesting may inuence the bone growth of donor sites, or for patients who have a phobia about bone harvesting, or when autogenous bone harvesting is contraindicated. As was shown for the rst time in this study, the double-step TDDO technique can achieve a satisfactory shape and

function in the reconstruction of angled mandibular defect involving ramus, angle and body. This technique is an option for the reconstruction of large angled mandibular defects when patients are prudently selected.
Funding

This study was supported by Technological Project Fund of Zhejiang Province of China (No. 2007C33012).
Competing Interests

None declared
Ethical Approval

Not required
References
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