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TMJ replacement utilizing patient-fitted TMJ TJR devices in a re-ankylosis child
Journal of Cranio-Maxillo-Facial Surgery 44 (2016) 493e499
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Case report
TMJ replacement utilizing patient-fitted TMJ TJR devices in a re-ankylosis child Piero Cascone a, Emanuela Basile a, Diletta Angeletti a, *, Valentino Vellone a, Valerio Ramieri a, PECRAM Study Group1 a
di Roma, Via del Policlinico, Roma, Italy Maxillo-Facial Surgery Dept, “Sapienza” Universita
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 6 October 2014 Accepted 16 June 2015 Available online 23 June 2015
Temporomandibular joint (TMJ) ankylosis is a pathological condition characterized by articular bony or fibrous tissue fusion. TMJ ankylosis developing during childhood can lead to growth complications because of the loss of mandibular function. Hard and soft autogenous tissue grafting has been used for TMJ reconstruction in the growing patient. However, in cases where autogenous tissue grafts fail either due to unpredictable growth or ankylosis, total alloplastic temporomandibular joint replacement (TMJ TJR) can provide a viable option. The case of a 7-year old female suffering from recurrent bilateral TMJ ankylosis resulting from birth trauma, and with concomitant obstructive sleep apnea syndrome (OSAS) is presented. Due to prior surgical and autogenous graft failures, the decision was made to complete her joint reconstructions utilizing patient-fitted TMJ prostheses. Questions have been raised about the longevity of TMJ TJR devices as well as their lack of growth potential, but children with TMJ ankylosis do not have condyle-related growth potential and that replacing failed autogenous tissue graft material with more autogenous tissue will result in the same adverse outcomes. Therefore, in growing patients with recurrent TMJ ankylosis and/or failed autogenous tissue grafts, there may be a role for TMJ TJR. © 2015 Published by Elsevier Ltd on behalf of European Association for Cranio-Maxillo-Facial Surgery.
Keywords: TMJ ankylosis TMJ bilateral ankylosis Growing patients TMJ ankylosis Total joint replacement in children TMJ TJR OSAS
1. Introduction Temporomandibular joint (TMJ) ankylosis is a pathological condition characterized by articular bony or fibrous tissue fusion. It can cause a dentofacial deformity resulting limitation of mandibular function, malocclusion, retrognathia, mandibular asymmetry, maxillary cant, malnutrition and poor oral hygiene (Zhang et al., 2012; Rowe, 1982). When TMJ ankylosis occurs during facial growth, it results in an alteration of entire maxillofacial complex. Moreover, mandibular hypomobility and subsequent retrognathia can produce a oropharyngeal airspace narrowing leading to the signs and symptoms of obstructive sleep apnea syndrome (OSAS) (Guruprasad and Hemavathy, 2012). A variety of techniques, with varying success rates for the management of TMJ ankylosis in both adults and children, have been reported (Guven, 2008). However, none has
1 A. Giancotti, M. Castori, J. Lenzi, L. Manganaro, P. Papoff, P. Pizzuti, A. Polimeni, M. Roggini, L. Tarani, A. Silvestri. * Corresponding author. Via N. Cocco, Rieti, Italy. Tel.: þ39 3200925175. E-mail address: [email protected] (D. Angeletti).
been reported consistently successful, illustrating the difficulty finding a completely satisfactory method for the management of TMJ ankylosis. Autogenous tissue grafting has been for TMJ reconstruction in the adult and growing patient. However, in cases where autogenous tissue grafts fail either due to unpredictable growth or ankylosis, total alloplastic temporomandibular joint replacement (TMJ TJR) can provide a viable option; as well as for the multiply operated patients with distorted TMJ anatomy (Egemen et al., 2012). The beneficial outcomes of TMJ TJR reported in adults lead to the consideration of TMJ TJR in the management of severe and recurrent ankylosis in children. Questions have been raised about the longevity of TMJ TJR devices as well as their lack of growth potential, but children with TMJ ankylosis do not have condylerelated growth potential and that replacing failed autogenous graft material with more autogenous tissue will result in the same adverse outcome. Therefore, in growing patients with recurrent TMJ ankylosis and/or failed autogenous tissue grafts, there may be a role for TMJ TJR.
http://dx.doi.org/10.1016/j.jcms.2015.06.018 1010-5182/© 2015 Published by Elsevier Ltd on behalf of European Association for Cranio-Maxillo-Facial Surgery.
494
P. Cascone et al. / Journal of Cranio-Maxillo-Facial Surgery 44 (2016) 493e499
This paper reports the case of a 7-year old female suffering from recurrent bilateral TMJ ankylosis resulting from birth trauma, and with concomitant obstructive sleep apnea syndrome (OSAS) managed with the use of patient-fitted TMJ TJR devices.
developed recurrence of bilateral TMJ ankylosis and significant reduction of MIO to 10 mm (Figs. 3 and 4a, b). At this point, a multi-step management plan was developed to implant bilateral patient-fitted TMJ TJR devices (TMJ Concepts, Ventura, CA, USA):
2. Case report
1. Bilateral TMJ gap arthroplasties and coronoidectomies with insertion of temporary silicone rubber spacers 2. Craniomandibular distraction to increase posterior vertical mandibular height, expand tissue and improve sagittal projection of the mandible 3. Bilateral patient-fitted TMJ TJR device design, manufacture and implantation
The patient presented for evaluation of her bilateral TMJ ankylosis to the Maxillo-Facial Surgery Department at the Sapienza di Roma at 5 months of age. She had a tracheostomy that Universita was performed at another hospital due to respiratory distress resulting from her severe mandibular micrognathia. Her past medical history obtained from her mother was significant in that allegedly at 35-weeks gestation, during Caesarean delivery, she was dropped sustaining undiagnosed bilateral mandibular condyle fractures. CT scans made at the initial visit revealed bilateral TMJ ankylosis and a compromised oropharyngeal airway space. Her micrognathia resulted in posterior displacement of the tongue, reduction of the oropharyngeal airway, obstruction and severe respiratory distress necessitating the aforementioned tracheostomy (Fig. 1a, b). At age 5 months, the patient underwent surgery to manage her bilateral TMJ ankylosis. The surgical plan involved gap arthroplasty, the interposition of a temporalis muscle flap, and mandibular advancement using external distraction devices (Cascone et al., 2014) (Fig. 2). The surgery was performed through a preauricular incision with temporal extension. The ankylotic bone mass was removed and the gap filled with a temporalis muscle/fascia flap rotated under the zygomatic arch and anchored with trans-osseous sutures to the anteromedial portion of the condyle. An osteotomy of each mandibular body was then performed and the external distractors applied. The mandible was advanced 18 mm over a period of 20 days and subsequently the tracheostomy was removed. Within a few days after surgery, the patient demonstrated an increased maximum incisal opening (MIO) from 1 mm to 10 mm and increased oropharyngeal airway space with no clinical signs or symptoms of OSAS. At age 4, despite normal maxillary growth, due to recurrence of the bilateral TMJ ankylosis, she demonstrated significantly diminished mandibular growth. Her MIO was 20 mm and a polysomnographic examination revealed severe OSAS. Further mandibular distraction was carried out with an internal device using a distraction rate of 1 mm daily. The mandible required 60 days for consolidation. A mandibular advancement of 20 mm with a concomitant increase in her oropharyngeal airway space was achieved. At age 6, a new polysomnographic examination revealed moderate OSAS (Respiratory Disturbance Index 5,1; SaO2 97,7). She continued to demonstrate severe mandibular retrusion and
2.1. Surgical technique At age 7, she underwent the first stage of a two-staged TMJ TJR protocol. A tracheostomy was performed to improve the OSAS management during this protocol. Stage 1 involved bilateral gap arthroplasties and coronoidectomies with the interposition of fitted silicone rubber spacers. These spacers were placed to maintain posterior mandibular vertical height and avoid the ingrowth of fibrous connective tissue between the first and second stages of the protocol. After Stage 1, a 2-piece (maxilla/mandible) stereolithic (SL) model (ProtoMED, Arvada, CO, USA) was developed from a protocol CT. The mandibular component of the 2-piece SL model was repositioned with the mandible overcorrected in a Class III occlusal in order to
Fig. 2. Patient after first surgery.
Fig. 1. CT scan performed at 5 months of age. A) Left lateral view. B) Right lateral view.
P. Cascone et al. / Journal of Cranio-Maxillo-Facial Surgery 44 (2016) 493e499
follow the maxillary growth. The SL model was used to design and manufacture the patient-fitted TMJ TJR devices (Fig. 5a, b). To achieve a counter-clockwise rotation of the mandible to increase the mandibular posterior height, expand the overlying soft tissue and improving the sagittal projection of the mandible, cranio-mandibular distractors were positioned (KLS Martin, Jacksonville, FL, USA). A total of 10 mm of projection was gained (Fig. 6a, b). Prior to Stage 2 surgery, an intermediate splint was designed to the planned maxillomandibular relationship. The patient then underwent Stage 2 surgery, bilateral TMJ TJR with patient-fitted devices (Fig. 7a, b). Two days post-operatively, the tracheostomy was removed and the patient started physiotherapy with active and passive exercises with a mandibular functional device (Therabite, Athos Medical, West Allies, WI, USA) in order to re-gain and maintain mandibular range of motion. Her MIO improved from 10 to 31 mm (Linsen et al., 2013; Guarda-Nardini et al., 2008). The 1 year post-operative follow up demonstrated a significant improvement of the patency of the upper airway space and good mandibular growth as well as a stable MIO of 21 mm (Figs. 8a, b, c and 9a, b, c). 3. Discussion The development of TMJ ankylosis during childhood presents significant mandibular growth disturbance problems for the patient and for those who attempt to manage them. The consequence TMJ ankylosis during maxillomandibular growth is the loss of the secondary growth potential of the condyle. Moreover normal masticatory muscular activity is affected resulting in loss of mandibular function and consequent appositional bone growth (Moss and Rankow, 1968). Since the essential life functions of mastication, speech, airway support and deglutition are supported from TMJ morphology and function, TMJ ankylosis in children leads to greater consequences than just decreased mandibular range of motion. The resulting altered growth and development of the maxillomandibular complex (El-Sheikh and Medra, 1997), malocclusion and airway obstruction lead to micrognathia and OSAS (Andrade et al., 2012; Li
Fig. 3. 6 years old patient, right side view.
495
et al., 2012; Cascone et al., 2013). Further, the retruded position of the mandible and the resulting micrognathia displace the tongue posteriorly thereby reducing the oropharyngeal airway space. Left untreated, many of these affected children would suffer significant complications, including failure to thrive due to lack of proper diet, chronic hypoxemia, neurological disorders, and rarely, cor pulmonale (Chang and Chae, 2010). Costochondral grafts have been reported as the “Gold Standard” for TMJ reconstruction in the growing patient (Ware and Taylor, 1966a,b; Ware and Brown, 1981; Poswillo, 1987; MacIntosh, 1985; MacIntosh, 2000). Theoretically, the costochondral grafts will “grow with the patient; ” however, often this so-called “growth potential” has been shown to be unpredictable or results in ankylosis, either due to allograft and/ or fixation failure, or due to the uncooperative nature of the young patient with post-reconstruction physical therapy (MacIntosh and Henny, 1977; Tasanen and Leikomma, 1977; Smith and Wolford, 1985; Obeid et al., 1988; Ware and Taylor, 1966a,b; Ware, 1970; Marx and Kretzschmar, 1983; Heffez and Doku, 1984; Politis et al., 1987; Lindqvist et al., 1988; Mulliken et al., 1989; Munro et al., 1989; Peltom€ aki and Isotupa, 1991; Fukuta et al., 1992; Guyuron and Lasa, 1992; Link et al., 1993; Samman et al., 1995) Recent studies have even questioned the necessity for using a cartilaginous graft to restore and maintain mandibular growth (Ellis et al., 2002; Guyot et al., 2004). Long-term reports of mandibular growth in children whose TMJs were reconstructed with costochondral grafts reveals the excessive growth on the treated side occurred in 54% of the 72 cases examined, and equal growth with the opposite side occurred in only 38% of the cases (Marx, 1992; Perrot et al., 1994; Svensson and Adell, 1998; Ross, 1999; Ko et al., 1999). Further, €a €ki et al. in investigations of mandibular growth after Peltoma costochondral grafting supported previous experiments with regard to the inability of the graft to adapt to the growth velocity of €ki et al., 2002; Peltoma €€ the new environment (Peltom€ aa aki and €nning, 1991; Peltom€ €ki, 1992). If autogenous grafts fail to Ro aa incorporate into the host bone, fail to grow, grow horizontally rather than vertically, or more commonly become ankylosed; the affected child require re-operation to either to debride the joint(s), re-place the joint(s) with another allograft(s), or has to wait until growth ceases to have another autogenous graft and/or orthognathic surgery and/or distraction osteogenesis procedures. Under these circumstances the child basically becomes disabled e unable to eat foods they and their peers crave because they cannot open their mouths wide enough, often they are not allowed participation in sports and games for fear they will sustain further injury or be unable to be resuscitated because the child cannot open their mouth. These and other issues can lead to social ostracization, depression and very poor life quality (Mercuri and Swift, 2009). The introduction of alloplastic TJR has improved the quality of life for many adult orthopedic and TMJ patients with unsalvageable functional and anatomical joint pathology (Mercuri and GiobbeHurder, 2004; Mercuri et al., 2007; Mercuri, 2012a). However, these devices, because they are a biomechanical rather than biological nature, have a finite life-span making them an unattractive alternative in the growing patient. Taking into consideration the information available for autogenous tissue grafting, total excision of the ankylotic segment and the use of alloplastic TMJ TJR may be an option in children with reankylosis of the TMJ (Mercuri, 2009; Westermark et al., 2011). Concern has been raised about the longevity of TMJ TJR devices and their lack of growth potential per se (Zhang et al., 2011). In spite of these potential drawbacks, children with TMJ ankylosis have no
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Fig. 4. CT scan performed at 6 years of age. A) Left lateral view. B) Right lateral view.
Fig. 5. CT scan performed with the silicone rubber spacers. A) Left lateral view. B) Right lateral view.
Fig. 6. CT scan performed with the cranio-mandibular distractors. A) Left lateral view. B) Right lateral view.
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Fig. 7. Prosthesis models. A) Left side. B) Right side.
condylar related growth potential and never will. Management of any eventual maxilla-mandibular growth discrepancies after TMJ TJR in such cases has not been broached in the literature to date as there is only one report of such a case (Mercuri, 2012b). However, one would assume that in these cases, the whole device or one of the components there of could be replaced with one of appropriate dimensions, or a ramus orthognatic procedure could be considered
since the ramus components of TMJ TJR devices, especially patientfitted devices, are implanted posterior to the mandibular foramen. Osseodistraction could also be considered using the mandibular component as one of the legs of the distraction device. It must be remembered that most of the children reconstructed with an autogenous graft require at least one more maxillomandibular surgical procedure anyway.
Fig. 8. 1 year post-operative CT with the prosthesis. A) Left side view B) Frontal view C) Right side view.
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Fig. 9. 12 years old patient. A) Left side. B) Frontal view. C) Frontal view at opened mouth.
4. Conclusion Temporomandibular joint ankylosis is a devastating condition for the growing patients and a challenging management problem for the clinician. The important management principles for these patients are: resolve the patients airway obstruction and respiratory distress, provide and maintain a satisfactory postoperative mouth opening and jaw function long-term, prevent re-ankylosis, provide the functional environment to allow the mandible to grow based on Moss's functional Matrix Theory. Currently, TMJ TJR is not the primary choice for the management of TMJ ankylosis in growing patients, but in selected cases, especially re-ankylosis, it may be considered as a possible treatment. Competing interests None declared. Funding None. Ethical approval Not required. Patient consent Patient agreed to publish clinical pictures. Acknowledgments A special thanks to Louis G. Mercuri, DDS, MS (Clinical Consultant TMJ Concepts and Visiting Professor Department of Orthopedic Surgery Rush University Medical Center Chicago, IL) for his important help in patient-fitted TMJ prostheses surgical planning and in the reviewing and correction of our manuscript. References Andrade NN, Kalra R, Shetye SP: New protocol to prevent TMJ reankylosis and potentially life threatening complications in triad patients. Int J Oral Maxillofac Surg 41(12): 1495e1500, 2012 Cascone P, Gennaro P, Gabriele G, Ramieri V: Functional outcomes in bilateral temporomandibular joint ankylosis treated with stock prosthesis. J Craniofac Surg 24(2): e139ee141, 2013
Cascone P, Papoff P, Arangio P, Vellone V, Calafati V, Silvestri A: Fast and early mandibular osteodistraction (FEMOD) in severe Pierre Robin Sequence. J Craniomaxillofac Surg 42(7): 1364e1370, 2014 Chang SJ, Chae KY: Obstructive sleep apnea syndrome in children: epidemiology, pathophysiology, diagnosis and sequelae. Korean J Pediatr 53(10): 863e871, 2010 Egemen O, Ozkaya O, Filinte GT, Uscetin I, Akan M: Two-Stage total prosthetic reconstruction of temporomandibular joint in severe and recurrent ankylosis. J Craniofac Surg 23(5): e520ee524, 2012 El-Sheikh MM, Medra AM: Management of unilateral temporomandibular ankylosis associated with facial asymmetry. J Craniomaxillofac Surg 25(3): 109e115, 1997 Ellis E, Schneiderman ED, Carlson DS: Growth of the mandible after replacement of the mandibular condyle: an experimental investigation in Macaca mulatta. J Oral Maxillofac Surg 60: 1461, 2002 Fukuta K, Jackson IT, Topf JS: Facial lawn mower injury treated by a vascularized costochondral graft. J Oral Maxillofac Surg 50: 194, 1992 Guarda-Nardini L, Manfredini D, Ferronato G: Total temporomandibular joint replacement: a clinical case with a proposal for post-surgical rehabilitation. J Craniomaxillofac Surg 36(7): 403e409, 2008 Guruprasad Y, Hemavathy OR: Management of obstructive sleep apnea syndrome secondary to temporomandibular joint ankylosis by mandibular elongation using distraction osteogenesis. Contemp Clin Dent 3(1): 69e73, 2012 Guven O: A clinical study on temporomandibular joint ankylosis in children. J Craniofac Surg 19(5): 1263e1269, 2008 Guyot L, Richard O, Layoun W, Cheynet F, Bellot-Samson V, Chossegros C, et al: Long-term radiological findings following reconstruction of the condyle with fibular free flaps. J Craniomaxillofac Surg 32: 98, 2004 Guyuron B, Lasa CI: Unpredictable growth pattern of costochondral graft. Plast Reconstr Surg 90: 880, 1992 Heffez L, Doku C: The Goldenhar syndrome: diagnosis and early surgical management. Oral Surg 58: 2, 1984 Ko EW, Huang CS, Chen YR: Temporomandibular joint reconstruction in children using costochondral grafts. J Oral Maxillofac Surg 57: 789, 1999 Li J, Zhu S, Wang T, Luo E, Xiao L, Hu J: Staged treatment of temporomandibular joint ankylosis with micrognathia using mandibular osteodistraction and advancement genioplasty. J Oral Maxillofac Surg 70(12): 2884e2892, 2012 Lindqvist C, Jokinen J, Paukku P, Tasanen A: Adaption of autogenous costochondral grafts used for temporomandibular joint reconstruction. J Oral Maxillofac Surg 46: 465, 1988 Link JO, Hoffman DC, Laskin DM: Hyperplasia of a costochondral graft. J Oral Maxillofac Surg 51: 1392, 1993 Linsen SS, Reich RH, Teschke M: Maximum voluntary bite force in patients with alloplastic total TMJ replacementea prospective study. J Craniomaxillofac Surg 41(5): 423e428, 2013 MacIntosh RB, Henny FA: A spectrum of application of autogenous costochondral grafts. J Oral Maxillofac Surg 5: 257, 1977 MacIntosh RB: Current spectrum of costochondral grafting. In: Bell WH (ed.), Surgical correction of dentofacial deformities: new concepts, vol. 3. Philadelphia: WB Saunders, 355e410, 1985 MacIntosh RB: The use of autogenous tissue in temporomandibular joint reconstruction. J Oral Maxillofac Surg 58: 63, 2000 Marx RE, Kretzschmar DP: Autogenous costochondral grafting in growing children. Curr Adv Oral Surg 4: 124, 1983 Marx RE: The science and art of reconstructing the jaws and temporomandibular joints. In: Bell WH (ed.), Modern practice in orthognathic and reconstructive surgery, vol. 2. Philadelphia, PA: Saunders, 1448, 1992
P. Cascone et al. / Journal of Cranio-Maxillo-Facial Surgery 44 (2016) 493e499 Mercuri LG, Edibam NR, Giobbie-Hurder A: 14-Year follow-up of a patient fitted total temporomandibular joint reconstruction system. J Oral Maxillofac Surg 65: 1140, 2007 Mercuri LG, Giobbe-Hurder A: Long term outcomes after total alloplastic TMJ reconstruction following exposure to failed materials. J Oral Maxillofac Surg 62: 1088, 2004 Mercuri LG, Swift JQ: Considerations for the use of alloplastic temporomandibular joint replacement in the growing patient. J Oral Maxillofac Surg 67: 1979e1990, 2009 Mercuri LG: Alloplastic TMJ replacement. Rationale for custom devices. Int J Oral Maxillofac Surg 41: 1033e1040, 2012a Mercuri LG: Temporomandibular joint reconstruction. Alpha Omegan 102(2): 51e54, 2009 Mercuri LG: Alloplastic temporomandibular joint replacement: rationale for the use of custom devices. Int J Oral Maxillofac Surg 41(9): 1033e1040, 2012b Moss ML, Rankow RM: The role of the functional matrix in mandibular growth. Angle Orthod 38: 95, 1968 Mulliken JB, Ferraro NF, Vento AR: A retrospective analysis of growth of the constructed condyle-ramus in children with hemifacial microsomia. Cleft Palate J 26: 312, 1989 Munro IR, Phillips JH, Griffin G: Growth after construction of the temporomandibular joint in children with hemifacial microsomia. Cleft Palate J 26: 303, 1989 Obeid G, Gutterman SA, Connole PW: Costochondral grafting in condylar replacement and mandibular reconstruction. J Oral Maxillofac Surg 48: 177, 1988 €€ € nning O: Interrelationship between size and tissue-separating Peltoma aki T, Ro potential of costochondral transplants. Eur J Orthod 13: 459, 1991 €€ €talo K, Ro € nning O: The effect of a unilateral cotchondral graft on Peltoma aki T, V€ aha the growth of the marmoset mandible. J Oral Maxillofac Surg 60: 1307, 2002 €€ Peltoma aki T: Growth of a costochondral graft in the rat temporomandibular joint. J Oral Maxillofac Surg 50: 851, 1992 €ki T, Isotupa K: The costochondral graft: a solution or a source of facial Peltoma asymmetry in growing children. A case report. Proc Finn Dent Soc 87: 167, 1991 Perrot DH, Umeda H, Kaban LB: Costochondral graft/reconstruction of the condyle/ ramus unit: long-term follow-up. Int J Oral Maxillofac Surg 23: 321, 1994
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Politis C, Jossion E, Bossuyt M: The use of costochondral grafts in arthroplasty of the temporomandibular joint. J Craniomaxillofac Surg 15: 345, 1987 Poswillo DE: Biological reconstruction of the mandibular condyle. Br L Oral Maxillofac Surg 25: 100, 1987 Ross RB: Costochondral grafts replacing the mandibular condyle. Cleft PalateCraniofac J 36: 334, 1999 Rowe NL: Ankylosis of the temporomandibular joint. J R Coll Surg Edinb 27(3): 167e173, 1982 Samman N, Cheung LK, Tideman H: Overgrowth of a costochondral graft in an adult male. Int J Oral Maxillofac Surg 24: 333, 1995 Smith BR, Wolford LM: Long-term growth in costochondral grafts in children; 1985, AAOMS Meeting Washington, DC Svensson A, Adell R: Costochondral grafts to replace mandibular condyles in juvenile chronic arthritis patients: long-term effects on facial growth. J Craniomaxillofac Surg 26: 275, 1998 Tasanen A, Leikomma H: Ankylosis of the TMJ of a child. Int J Oral Surg 6: 95, 1977 Ware WH, Brown SL: Growth center transplantation to replace mandibular condyles. J Maxillofac Surg 9: 50e58, 1981 Ware WH, Taylor RC: Cartilaginous growth centers transplanted to replace mandibular condyles in monkeys. J Oral Surg 24: 33e43, 1966a Ware WH, Taylor RC: Cartilaginous growth centers transplanted to replace mandibular condyles in monkeys. J Oral Surg 24: 33, 1966b Ware WH: Growth center transplantation in TMJ surgery. In: Walker RV (ed.), Transactions of the Third Congress of the International Association of Oral and Maxillofacial Surgeons; 1970, 148e157, 1970 n P, Aagaard E, Cornelius CP: The use of TMJ concepts prosWestermark A, Hede theses to reconstruct patients with major temporomandibular joint and mandibular defects. Int J Oral Maxillofac Surg 40: 487e496, 2011 Zhang X, Chen M, Wu Y, Wang B, Yang C: Management of temporomandibular joint ankylosis associated with mandibular asymmetry in infancy. J Craniofac Surg 22(4): 1316e1319, 2011 Zhang XH, Yang C, Fang B, Chen MJ, Wu Y, Wang BL: Simultaneous costochondral graft and distraction osteogenesis in unilateral TMJ ankylosis associated with mandibular retrognathia and asymmetry. J Craniofac Surg 23(3): 682e684, 2012
Contents lists available at ScienceDirect
Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com
Case report
TMJ replacement utilizing patient-fitted TMJ TJR devices in a re-ankylosis child Piero Cascone a, Emanuela Basile a, Diletta Angeletti a, *, Valentino Vellone a, Valerio Ramieri a, PECRAM Study Group1 a
di Roma, Via del Policlinico, Roma, Italy Maxillo-Facial Surgery Dept, “Sapienza” Universita
a r t i c l e i n f o
a b s t r a c t
Article history: Paper received 6 October 2014 Accepted 16 June 2015 Available online 23 June 2015
Temporomandibular joint (TMJ) ankylosis is a pathological condition characterized by articular bony or fibrous tissue fusion. TMJ ankylosis developing during childhood can lead to growth complications because of the loss of mandibular function. Hard and soft autogenous tissue grafting has been used for TMJ reconstruction in the growing patient. However, in cases where autogenous tissue grafts fail either due to unpredictable growth or ankylosis, total alloplastic temporomandibular joint replacement (TMJ TJR) can provide a viable option. The case of a 7-year old female suffering from recurrent bilateral TMJ ankylosis resulting from birth trauma, and with concomitant obstructive sleep apnea syndrome (OSAS) is presented. Due to prior surgical and autogenous graft failures, the decision was made to complete her joint reconstructions utilizing patient-fitted TMJ prostheses. Questions have been raised about the longevity of TMJ TJR devices as well as their lack of growth potential, but children with TMJ ankylosis do not have condyle-related growth potential and that replacing failed autogenous tissue graft material with more autogenous tissue will result in the same adverse outcomes. Therefore, in growing patients with recurrent TMJ ankylosis and/or failed autogenous tissue grafts, there may be a role for TMJ TJR. © 2015 Published by Elsevier Ltd on behalf of European Association for Cranio-Maxillo-Facial Surgery.
Keywords: TMJ ankylosis TMJ bilateral ankylosis Growing patients TMJ ankylosis Total joint replacement in children TMJ TJR OSAS
1. Introduction Temporomandibular joint (TMJ) ankylosis is a pathological condition characterized by articular bony or fibrous tissue fusion. It can cause a dentofacial deformity resulting limitation of mandibular function, malocclusion, retrognathia, mandibular asymmetry, maxillary cant, malnutrition and poor oral hygiene (Zhang et al., 2012; Rowe, 1982). When TMJ ankylosis occurs during facial growth, it results in an alteration of entire maxillofacial complex. Moreover, mandibular hypomobility and subsequent retrognathia can produce a oropharyngeal airspace narrowing leading to the signs and symptoms of obstructive sleep apnea syndrome (OSAS) (Guruprasad and Hemavathy, 2012). A variety of techniques, with varying success rates for the management of TMJ ankylosis in both adults and children, have been reported (Guven, 2008). However, none has
1 A. Giancotti, M. Castori, J. Lenzi, L. Manganaro, P. Papoff, P. Pizzuti, A. Polimeni, M. Roggini, L. Tarani, A. Silvestri. * Corresponding author. Via N. Cocco, Rieti, Italy. Tel.: þ39 3200925175. E-mail address: [email protected] (D. Angeletti).
been reported consistently successful, illustrating the difficulty finding a completely satisfactory method for the management of TMJ ankylosis. Autogenous tissue grafting has been for TMJ reconstruction in the adult and growing patient. However, in cases where autogenous tissue grafts fail either due to unpredictable growth or ankylosis, total alloplastic temporomandibular joint replacement (TMJ TJR) can provide a viable option; as well as for the multiply operated patients with distorted TMJ anatomy (Egemen et al., 2012). The beneficial outcomes of TMJ TJR reported in adults lead to the consideration of TMJ TJR in the management of severe and recurrent ankylosis in children. Questions have been raised about the longevity of TMJ TJR devices as well as their lack of growth potential, but children with TMJ ankylosis do not have condylerelated growth potential and that replacing failed autogenous graft material with more autogenous tissue will result in the same adverse outcome. Therefore, in growing patients with recurrent TMJ ankylosis and/or failed autogenous tissue grafts, there may be a role for TMJ TJR.
http://dx.doi.org/10.1016/j.jcms.2015.06.018 1010-5182/© 2015 Published by Elsevier Ltd on behalf of European Association for Cranio-Maxillo-Facial Surgery.
494
P. Cascone et al. / Journal of Cranio-Maxillo-Facial Surgery 44 (2016) 493e499
This paper reports the case of a 7-year old female suffering from recurrent bilateral TMJ ankylosis resulting from birth trauma, and with concomitant obstructive sleep apnea syndrome (OSAS) managed with the use of patient-fitted TMJ TJR devices.
developed recurrence of bilateral TMJ ankylosis and significant reduction of MIO to 10 mm (Figs. 3 and 4a, b). At this point, a multi-step management plan was developed to implant bilateral patient-fitted TMJ TJR devices (TMJ Concepts, Ventura, CA, USA):
2. Case report
1. Bilateral TMJ gap arthroplasties and coronoidectomies with insertion of temporary silicone rubber spacers 2. Craniomandibular distraction to increase posterior vertical mandibular height, expand tissue and improve sagittal projection of the mandible 3. Bilateral patient-fitted TMJ TJR device design, manufacture and implantation
The patient presented for evaluation of her bilateral TMJ ankylosis to the Maxillo-Facial Surgery Department at the Sapienza di Roma at 5 months of age. She had a tracheostomy that Universita was performed at another hospital due to respiratory distress resulting from her severe mandibular micrognathia. Her past medical history obtained from her mother was significant in that allegedly at 35-weeks gestation, during Caesarean delivery, she was dropped sustaining undiagnosed bilateral mandibular condyle fractures. CT scans made at the initial visit revealed bilateral TMJ ankylosis and a compromised oropharyngeal airway space. Her micrognathia resulted in posterior displacement of the tongue, reduction of the oropharyngeal airway, obstruction and severe respiratory distress necessitating the aforementioned tracheostomy (Fig. 1a, b). At age 5 months, the patient underwent surgery to manage her bilateral TMJ ankylosis. The surgical plan involved gap arthroplasty, the interposition of a temporalis muscle flap, and mandibular advancement using external distraction devices (Cascone et al., 2014) (Fig. 2). The surgery was performed through a preauricular incision with temporal extension. The ankylotic bone mass was removed and the gap filled with a temporalis muscle/fascia flap rotated under the zygomatic arch and anchored with trans-osseous sutures to the anteromedial portion of the condyle. An osteotomy of each mandibular body was then performed and the external distractors applied. The mandible was advanced 18 mm over a period of 20 days and subsequently the tracheostomy was removed. Within a few days after surgery, the patient demonstrated an increased maximum incisal opening (MIO) from 1 mm to 10 mm and increased oropharyngeal airway space with no clinical signs or symptoms of OSAS. At age 4, despite normal maxillary growth, due to recurrence of the bilateral TMJ ankylosis, she demonstrated significantly diminished mandibular growth. Her MIO was 20 mm and a polysomnographic examination revealed severe OSAS. Further mandibular distraction was carried out with an internal device using a distraction rate of 1 mm daily. The mandible required 60 days for consolidation. A mandibular advancement of 20 mm with a concomitant increase in her oropharyngeal airway space was achieved. At age 6, a new polysomnographic examination revealed moderate OSAS (Respiratory Disturbance Index 5,1; SaO2 97,7). She continued to demonstrate severe mandibular retrusion and
2.1. Surgical technique At age 7, she underwent the first stage of a two-staged TMJ TJR protocol. A tracheostomy was performed to improve the OSAS management during this protocol. Stage 1 involved bilateral gap arthroplasties and coronoidectomies with the interposition of fitted silicone rubber spacers. These spacers were placed to maintain posterior mandibular vertical height and avoid the ingrowth of fibrous connective tissue between the first and second stages of the protocol. After Stage 1, a 2-piece (maxilla/mandible) stereolithic (SL) model (ProtoMED, Arvada, CO, USA) was developed from a protocol CT. The mandibular component of the 2-piece SL model was repositioned with the mandible overcorrected in a Class III occlusal in order to
Fig. 2. Patient after first surgery.
Fig. 1. CT scan performed at 5 months of age. A) Left lateral view. B) Right lateral view.
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follow the maxillary growth. The SL model was used to design and manufacture the patient-fitted TMJ TJR devices (Fig. 5a, b). To achieve a counter-clockwise rotation of the mandible to increase the mandibular posterior height, expand the overlying soft tissue and improving the sagittal projection of the mandible, cranio-mandibular distractors were positioned (KLS Martin, Jacksonville, FL, USA). A total of 10 mm of projection was gained (Fig. 6a, b). Prior to Stage 2 surgery, an intermediate splint was designed to the planned maxillomandibular relationship. The patient then underwent Stage 2 surgery, bilateral TMJ TJR with patient-fitted devices (Fig. 7a, b). Two days post-operatively, the tracheostomy was removed and the patient started physiotherapy with active and passive exercises with a mandibular functional device (Therabite, Athos Medical, West Allies, WI, USA) in order to re-gain and maintain mandibular range of motion. Her MIO improved from 10 to 31 mm (Linsen et al., 2013; Guarda-Nardini et al., 2008). The 1 year post-operative follow up demonstrated a significant improvement of the patency of the upper airway space and good mandibular growth as well as a stable MIO of 21 mm (Figs. 8a, b, c and 9a, b, c). 3. Discussion The development of TMJ ankylosis during childhood presents significant mandibular growth disturbance problems for the patient and for those who attempt to manage them. The consequence TMJ ankylosis during maxillomandibular growth is the loss of the secondary growth potential of the condyle. Moreover normal masticatory muscular activity is affected resulting in loss of mandibular function and consequent appositional bone growth (Moss and Rankow, 1968). Since the essential life functions of mastication, speech, airway support and deglutition are supported from TMJ morphology and function, TMJ ankylosis in children leads to greater consequences than just decreased mandibular range of motion. The resulting altered growth and development of the maxillomandibular complex (El-Sheikh and Medra, 1997), malocclusion and airway obstruction lead to micrognathia and OSAS (Andrade et al., 2012; Li
Fig. 3. 6 years old patient, right side view.
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et al., 2012; Cascone et al., 2013). Further, the retruded position of the mandible and the resulting micrognathia displace the tongue posteriorly thereby reducing the oropharyngeal airway space. Left untreated, many of these affected children would suffer significant complications, including failure to thrive due to lack of proper diet, chronic hypoxemia, neurological disorders, and rarely, cor pulmonale (Chang and Chae, 2010). Costochondral grafts have been reported as the “Gold Standard” for TMJ reconstruction in the growing patient (Ware and Taylor, 1966a,b; Ware and Brown, 1981; Poswillo, 1987; MacIntosh, 1985; MacIntosh, 2000). Theoretically, the costochondral grafts will “grow with the patient; ” however, often this so-called “growth potential” has been shown to be unpredictable or results in ankylosis, either due to allograft and/ or fixation failure, or due to the uncooperative nature of the young patient with post-reconstruction physical therapy (MacIntosh and Henny, 1977; Tasanen and Leikomma, 1977; Smith and Wolford, 1985; Obeid et al., 1988; Ware and Taylor, 1966a,b; Ware, 1970; Marx and Kretzschmar, 1983; Heffez and Doku, 1984; Politis et al., 1987; Lindqvist et al., 1988; Mulliken et al., 1989; Munro et al., 1989; Peltom€ aki and Isotupa, 1991; Fukuta et al., 1992; Guyuron and Lasa, 1992; Link et al., 1993; Samman et al., 1995) Recent studies have even questioned the necessity for using a cartilaginous graft to restore and maintain mandibular growth (Ellis et al., 2002; Guyot et al., 2004). Long-term reports of mandibular growth in children whose TMJs were reconstructed with costochondral grafts reveals the excessive growth on the treated side occurred in 54% of the 72 cases examined, and equal growth with the opposite side occurred in only 38% of the cases (Marx, 1992; Perrot et al., 1994; Svensson and Adell, 1998; Ross, 1999; Ko et al., 1999). Further, €a €ki et al. in investigations of mandibular growth after Peltoma costochondral grafting supported previous experiments with regard to the inability of the graft to adapt to the growth velocity of €ki et al., 2002; Peltoma €€ the new environment (Peltom€ aa aki and €nning, 1991; Peltom€ €ki, 1992). If autogenous grafts fail to Ro aa incorporate into the host bone, fail to grow, grow horizontally rather than vertically, or more commonly become ankylosed; the affected child require re-operation to either to debride the joint(s), re-place the joint(s) with another allograft(s), or has to wait until growth ceases to have another autogenous graft and/or orthognathic surgery and/or distraction osteogenesis procedures. Under these circumstances the child basically becomes disabled e unable to eat foods they and their peers crave because they cannot open their mouths wide enough, often they are not allowed participation in sports and games for fear they will sustain further injury or be unable to be resuscitated because the child cannot open their mouth. These and other issues can lead to social ostracization, depression and very poor life quality (Mercuri and Swift, 2009). The introduction of alloplastic TJR has improved the quality of life for many adult orthopedic and TMJ patients with unsalvageable functional and anatomical joint pathology (Mercuri and GiobbeHurder, 2004; Mercuri et al., 2007; Mercuri, 2012a). However, these devices, because they are a biomechanical rather than biological nature, have a finite life-span making them an unattractive alternative in the growing patient. Taking into consideration the information available for autogenous tissue grafting, total excision of the ankylotic segment and the use of alloplastic TMJ TJR may be an option in children with reankylosis of the TMJ (Mercuri, 2009; Westermark et al., 2011). Concern has been raised about the longevity of TMJ TJR devices and their lack of growth potential per se (Zhang et al., 2011). In spite of these potential drawbacks, children with TMJ ankylosis have no
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Fig. 4. CT scan performed at 6 years of age. A) Left lateral view. B) Right lateral view.
Fig. 5. CT scan performed with the silicone rubber spacers. A) Left lateral view. B) Right lateral view.
Fig. 6. CT scan performed with the cranio-mandibular distractors. A) Left lateral view. B) Right lateral view.
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Fig. 7. Prosthesis models. A) Left side. B) Right side.
condylar related growth potential and never will. Management of any eventual maxilla-mandibular growth discrepancies after TMJ TJR in such cases has not been broached in the literature to date as there is only one report of such a case (Mercuri, 2012b). However, one would assume that in these cases, the whole device or one of the components there of could be replaced with one of appropriate dimensions, or a ramus orthognatic procedure could be considered
since the ramus components of TMJ TJR devices, especially patientfitted devices, are implanted posterior to the mandibular foramen. Osseodistraction could also be considered using the mandibular component as one of the legs of the distraction device. It must be remembered that most of the children reconstructed with an autogenous graft require at least one more maxillomandibular surgical procedure anyway.
Fig. 8. 1 year post-operative CT with the prosthesis. A) Left side view B) Frontal view C) Right side view.
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Fig. 9. 12 years old patient. A) Left side. B) Frontal view. C) Frontal view at opened mouth.
4. Conclusion Temporomandibular joint ankylosis is a devastating condition for the growing patients and a challenging management problem for the clinician. The important management principles for these patients are: resolve the patients airway obstruction and respiratory distress, provide and maintain a satisfactory postoperative mouth opening and jaw function long-term, prevent re-ankylosis, provide the functional environment to allow the mandible to grow based on Moss's functional Matrix Theory. Currently, TMJ TJR is not the primary choice for the management of TMJ ankylosis in growing patients, but in selected cases, especially re-ankylosis, it may be considered as a possible treatment. Competing interests None declared. Funding None. Ethical approval Not required. Patient consent Patient agreed to publish clinical pictures. Acknowledgments A special thanks to Louis G. Mercuri, DDS, MS (Clinical Consultant TMJ Concepts and Visiting Professor Department of Orthopedic Surgery Rush University Medical Center Chicago, IL) for his important help in patient-fitted TMJ prostheses surgical planning and in the reviewing and correction of our manuscript. References Andrade NN, Kalra R, Shetye SP: New protocol to prevent TMJ reankylosis and potentially life threatening complications in triad patients. Int J Oral Maxillofac Surg 41(12): 1495e1500, 2012 Cascone P, Gennaro P, Gabriele G, Ramieri V: Functional outcomes in bilateral temporomandibular joint ankylosis treated with stock prosthesis. J Craniofac Surg 24(2): e139ee141, 2013
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