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Int. J. Oral Maxillofac. Surg. 2013; xxx: xxx–xxx http://dx.doi.org/10.1016/j.ijom.2013.07.743, available online at http://www.sciencedirect.com
Case Report Craniofacial Anomalies
Unusual costochondral bone graft complication Nathalie Tabchouri, Natacha Kadlub, Patrick A. Diner, Arnaud Picard: Unusual costochondral bone graft complication. Int. J. Oral Maxillofac. Surg. 2013; xxx: xxx– xxx. # 2013 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Abstract. In hemifacial microsomia, patients with severely hypoplastic mandibles (Pruzansky type III) require replacement of the ramus and condyle unit. Common complications of this procedure include graft fracture and overgrowth of the graft. An uncommon case of osteolysis of the costochondral graft with osteitis of the middle cranial fossa is reported herein. To our knowledge, no such case has been reported in the literature previously. The aim of this report is to present the only known case and to discuss the contributing factors.
Introduction
In hemifacial microsomia, patients with severely hypoplastic mandibles (Pruzansky type III) require replacement of the ramus and condyle unit. A costochondral graft is the best-known option for reconstruction of the ramus and temporomandibular joint (TMJ), because of further growth of the graft1. The most common complications observed are graft fracture, rib graft failure, and under or overgrowth of the graft. Osteolysis of the middle cranial fossa by a costochondral graft is an unusual event, with no case reported in the literature to our knowledge. We describe herein the case of a 6-year-old girl with otomandibular dysplasia who presented with osteolysis of the middle cranial fossa following a costochondral graft. The cause of this complication is discussed with reference to the literature. Case report
A 6-year-old girl was referred to the department of maxillofacial and plastic 0901-5027/000001+04 $36.00/0
surgery for treatment of left Pruzansky III otomandibular dysplasia type (Fig. 1). Clinical evaluation showed a deviated chin, an occlusal cant, a scissor bite, and a maximal inter-incisal mouth opening of 27 mm. An orthodontic device was used preoperatively to counter the lateral deviation and to smoothly elongate the muscles to prevent postoperative contraction. Stereolithographic reconstruction (OBL, Chaˆtillon, France) was used to evaluate the length, the outward angulation of the distal segment of the bone, and the angulation necessary for the reconstruction. An under-correction was planned in order to prevent postoperative trismus and temporal fossa conflict. Ramus reconstruction with a costochondral graft was performed at the age of 6 years. A right seventh rib graft was harvested subperichondrally. The choice of chest side was guided by the further need for a left ear reconstruction. The perichondrium of the costochondral junction was preserved to
Nathalie Tabchouria,b, Natacha Kadluba,b,c,d, Patrick A.Dinera,b, Arnaud Picarda,b,d,e a
Service de Chirurgie Maxillo-faciale et Plastique Pe´diatrique, APHP, Hoˆpital Necker Enfants Malades, Paris, France; bCentre de Re´fe´rence des Maladie Rares de la Face et de la Cavite´ Buccale, APHP, Hoˆpital Necker Enfants Malades, Paris, France; cFaculte´ de Me´decine Paris Descartes, Universite´ Paris 5, Paris, France; dCentre de Recherche des Cordeliers, INSERM, Paris, France; eFaculte´ de Me´decine Pierre et Marie Curie, Universite´ Paris 6, Paris, France
Keywords: Hemifacial microsomia; Costochondral bone graft; Otomandibular dysplasia; Middle crania fossa perforation. Accepted for publication 16 July 2013
maintain bone–cartilage connection for further chest growth. The length of the graft was 6.5 cm, including 13 mm of cartilage. According to the stereolithographic analysis, the graft length had been undercorrected by 10 mm. Intravenous prophylactic antibiotics were administered during and for 5 days after the procedure. The bone graft was performed through an intraoral approach. A lateral vestibular incision was made along the external oblique line to the first left lateral incisors. A subperiosteal dissection was performed to expose the horizontal branch of the mandible. A tunnel was made towards the glenoid fossa with a non-traumatic fingertip dissection. The graft was then set in place with maximal bone contact between the graft and the mandibular horizontal branch (onlay technique). The recipient mandibular bone was abraded to prevent non-union. Osteosynthesis was performed with a 6-hole 2.0mm titanium plate (Fig. 2). At the end of the surgery, the surgeon did not note any conflict between the middle cranial fossa and
# 2013 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Tabchouri N, et al. Unusual costochondral bone graft complication, Int J Oral Maxillofac Surg (2013), http://dx.doi.org/10.1016/j.ijom.2013.07.743
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Fig. 3. (A) CT scan showing osteolysis of the middle cranial fossa; the graft is lifted into the medial cranial fossa without dura abscess or perforation. (B) CT scan, coronal view, showing an intact cranial fossa, before the second reconstructive surgery.
Fig. 1. Preoperative photographs: (A) frontal photograph showing the chin point deviation; (B) CT scan, three-dimensional reconstruction, showing the severe mandibular hypoplasia (Pruzansky III); (C) CT scan, coronal view, showing an intact cranial base.
Fig. 2. X-ray at 1 week postoperative showing the costochondral graft.
the reconstructed ramus. Immediate postoperative evaluation showed restored facial symmetry (chin point in the facial midline) and a maximal inter-incisal opening of up to 30 mm. The surgical approach was closed in two interrupted suture layers. No maxillomandibular fixation was applied, but a bite guide was applied to achieve occlusal contacts. On day 7, a minimal mucosa wound dehiscence was noted. Surgical exploration did not show any graft exposure, and closure was performed with a bioresorbable suture. At week 4, the patient presented with left mandibular localized cellulitis without trismus or scar disunion. On taking a history, the parents reported a cough; a physical examination was unremarkable showing a normal external auditory meatus. The patient was admitted and administered intravenous amoxicillin–clavulanic acid for 7 days. A craniofacial computed tomography (CT) scan revealed partial costochondral graft resorption and middle cranial fossa osteolysis without any plate
fracture or screw loosening. The graft was lifted up into the middle cranial fossa without dehiscence of the dura. No intracranial pneumatocele, abscess, or haematoma was noted (Fig. 3A). A multidisciplinary staff meeting involving maxillofacial surgeons, bacteriologists, paediatricians, and neurosurgeons led to the decision to remove the graft. Surgical exploration showed lost screws, osteitis of the graft, and total loosening of the cartilage. Pathological examination confirmed osteitis. Bacteriological cultures of the graft revealed a Streptococcus species infection sensitive to the antibiotics administered. Intravenous antibiotics were continued for 10 days, followed by a 6-week course of oral amoxicillin. Close clinical and CT scan follow-up showed resolution of the infection and middle cranial base reconstruction. A year later, a multidisciplinary staff meeting considered a new reconstruction. Preoperatively, bacteriological samples (skin, nose, and throat) did not show any pathological agent. A preoperative CT scan showed complete resolution of the infection and an intact cranial base (Fig. 3B). Prophylactic preoperative oral antibiotics (amoxicillin–clavulanic acid)
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Unusual costochondral bone graft complication
Fig. 4. Photograph showing the patient 1 year after the second reconstructive surgery.
were administered. The patient then underwent the same surgical procedure as described previously. The rib graft length was shortened (4 cm, including 20 mm of cartilage) to avoid any cranial base conflict. Postoperatively, a 7-week course of antibiotics (in accordance with the Streptococcus species antibiogram spectrum) was administered intravenously. No infection or osteitis occurred. Follow-up over the following 1 year was uneventful (Fig. 4). Discussion
Management of the reconstruction of congenital conditions such as hemifacial microsomia depends on the severity of the bone defect. Hemifacial microsomia is known for its wide spectrum, hence the multiple therapeutic options. Pruzansky type III is the most severe hemifacial microsomia subtype, characterized by an absent TMJ and an absent or severely hypoplastic ramus. Hypoplastic mandible reconstruction is dependent on the degree of ramus and condyle deficiency. Distraction osteogenesis or a bone graft may be proposed. Patients with Pruzansky type III hemifacial microsomia require a bone graft to restore the ramus and condyle unit2,3. Currently, the costochondral graft is the most accepted technique2,4. Kaban et al.2 recommend early childhood reconstruction in mixed dentition due to the potential for midface vertical growth as deciduous teeth are shed and permanent teeth erupt. The benefits of early reconstruction include improved mandibular and maxillary growth, improved development of body image, and reduced secondary maxilla deformities2. To date, no previous article has described this complication in the reconstruction of a
hemifacial microsomia. Postoperative erosion of the skull base has been reported by Lindqvist et al.5; however the skull base erosion concerned condyle reconstruction with metallic implants, without infectious complications5. Post-traumatic mandible condyle dislocation into the middle cranial fossa had also been described6–8. In these cases, again, intracranial migration was due to a traumatic mechanism without any infectious process. In our case, the middle cranial erosion was due to infection and secondary osteitis. Lypka et al.9 reported a case of middle cranial fossa perforation after autologous TMJ reconstruction. In this case, the patient suffered from a rhabdomyosarcoma with intracranial extension. The patient had been treated with radiotherapy and had developed TMJ ankylosis, which was treated with ankylosis release and costochondral graft reconstruction. The patient presented with an immediate postoperative infection and a delayed perforation of the middle cranial fossa. Unfortunately, the authors did not describe their surgical technique, the bacteriological pathological agent, or the pathological examination of the graft. The authors assume radiotherapy and an early postoperative infection were the likely contributing factors. In our case, the patient did not present with any such risk factors. We assume the postoperative infection may have been due to a haematoma and intraoral bacteriological contamination. Our surgical technique differs from that reported in the literature3 with regard to the surgical approach and graft fixation. Thaller and Bradley3 and Wan et al.4 recommend a 3 cm submental incision, fingertip dissection towards the glenoid fossa, and lag screw fixation. Controversy may exist regarding the surgical approach. We chose an intraoral approach to avoid facial nerve palsy and a cutaneous scar. Moreover, this approach allowed for good exposure of the surgical field. It should be noted that an intraoral approach may increase graft bacterial contamination, wound dehiscence, and graft exposure. However in our case, the osteitis was due to a Streptococcus species that is an intraoral non-pathogenic bacteriological agent. To minimize the infection risk, antibiotics were administered intravenously postoperatively and pursued orally. A prospective controlled study of intraoral bone grafting showed an increase in infectious complications after intraoral bone grafts without antibiotic prophylaxis10. No guidelines have been established for
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intraoral bone grafts. In order to limit the postoperative discomfort and prevent germ inoculation, no drain was placed in our case. We conjecture that the absence of a drain may have provoked formation of a haematoma and thereby a secondary bone graft infection. However, a fingertip dissection was used to make a tunnel towards the cranial base, which is less traumatic than other techniques and should reduce the risk of haematoma formation. To our knowledge, this is the first case of early middle cranial base osteitis secondary to a costochondral graft in a hemifacial microsomia patient reported in the literature. This complication did not contraindicate further reconstruction. Funding
No funding source for this research. Competing interests
No conflict of interest. Ethical approval
Not required. Patient consent
Patient consent was obtained to publish the clinical photographs. References 1. Kaban LB, Bouchard C, Troulis MJ. A protocol for management of temporomandibular joint ankylosis in children. J Oral Maxillofac Surg 2009;67:1966–78. 2. Kaban LB, Padwa BL, Mulliken JB. Surgical correction of mandibular hypoplasia in hemifacial microsomia: the case for treatment in early childhood. J Oral Maxillofac Surg 1998;56:628–38. 3. Thaller SR, Bradley JP. Craniofacial surgery. New York: Informa Healthcare; 2008. 4. Wan DC, Taub PJ, Allam KA, Perry A, Tabit CJ, Kawamoto HK, et al. Distraction osteogenesis of costocartilaginous rib grafts and treatment algorithm for severely hypoplastic mandibles. Plast Reconstr Surg 2011;127: 2005–13. 5. Lindqvist C, So¨derholm AL, Hallikainen D, Sjo¨vall L. Erosion and heterotopic bone formation after alloplastic temporomandibular joint reconstruction. J Oral Maxillofac Surg 1992;50:942–9. 6. Van der Linden WJ. Dislocation of the mandibular condyle into the middle cranial fossa: report of a case with 5 year CT follow-up. Int J Oral Maxillofac Surg 2003;32:215–8.
Please cite this article in press as: Tabchouri N, et al. Unusual costochondral bone graft complication, Int J Oral Maxillofac Surg (2013), http://dx.doi.org/10.1016/j.ijom.2013.07.743
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7. Cillo JE, Sinn DP, Ellis III E. Traumatic dislocation of the mandibular condyle into the middle cranial fossa treated with immediate reconstruction: a case report. J Oral Maxillofac Surg 2005;63:859–65. 8. Rosa VL, Guimaraes AS, Marie SK. Intrusion of the mandibular condyle into the middle cranial fossa: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:e4–7.
9. Lypka M, Urata M, Hammoudeh J. Delayed middle cranial fossa perforation after autologous temporomandibular joint reconstruction. J Oral Maxillofac Surg 2011;69:1613–6. 10. Lindeboom JA, van den Akker HP. A prospective placebo-controlled double-blind trial of antibiotic prophylaxis in intraoral bone grafting procedures: a pilot study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;96:669–72.
Corresponding author at: Service de Chirurgie Maxillo-faciale et Plastique Hoˆpital Necker Enfants Malades 149 rue de Se`vres 75015 Paris France Tel.: +33 1 44 49 43 15 E-mail:
[email protected]
Please cite this article in press as: Tabchouri N, et al. Unusual costochondral bone graft complication, Int J Oral Maxillofac Surg (2013), http://dx.doi.org/10.1016/j.ijom.2013.07.743