The Cleft Palate–Craniofacial Journal 52(3) pp. 373–376 May 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association

CASE REPORT Carotid Artery Dissection and Stroke Complicating Treatment of Postmandibular Distraction Ankylosis: A Case Report Sarah A. Arnspiger, B.S., John M. Felder III, M.D., Benjamin C. Wood, M.D., Gary F. Rogers, M.D., J.D., M.B.A., Albert K. Oh, M.D. Mandibular distraction osteogenesis is an increasingly accepted treatment option for severe upper airway obstruction in grade 3 Robin sequence. Complications are rarely reported but can include fracture, pin dislodgement, tooth bud damage, and temporomandibular joint ankylosis. Operative correction of these complications can carry inherent risks of their own. We present a patient who incurred carotid artery dissection and stroke after release of postdistraction coronoid-zygomatic ankylosis for the treatment of mandibular micrognathia. KEY WORDS:

distraction osteogenesis, mandibular distraction, mandibular pseudoankylosis, micrognathia, Robin sequence, stroke, surgical complications, Treacher Collins syndrome

Since its initial descriptions (Snyder et al., 1973; McCarthy et al., 1992), mandibular distraction osteogenesis (MDO) has proven to be an effective and relatively safe method for management of severe airway compromise associated with Robin sequence. Nevertheless, this technique is not without complications. A recent report found an overall complication rate of 20.5% to 35.6%, the most common of which was relapse or regression observed in 64.8% of patients (Master et al., 2010). Relapse was more common in younger patients, and this has led some surgeons to wait until skeletal maturity in patients without airway issues (Van Strijen et al., 2004; Gursoy et al., 2008). Other complications include tooth injury (22.5%; Mofid et al., 2001; Swennen et al., 2001; Freitas et al., 2008), hypertrophic scarring (15.6%), nerve injury (11.4%; McCarthy, 1999; Master et al., 2010), infection (9.5%), inappropriate distraction vector (8.8%; Shetye et al., 2009), device failure (7.9%; Uckan et al., 2006), and failure of consolidation (2.4%; McCarthy et al., 2001; McCarthy et al., 2002). Injury to the temporomandibular joint (TMJ) following distraction is uncommon and is most frequently associated with inappropriate distraction vector or placement of the

osteotomy cephalad to the sigmoid notch (Master et al., 2010). We present a patient with Treacher Collins syndrome and severe Robin sequence who developed TMJ ankylosis following MDO at an outside institution. Correction of the ankylosis at our institution improved mandibular excursion but led to a carotid artery dissection and stroke. This case highlights a rare complication of MDO and the potentially life-threatening consequence of TMJ ankylosis correction. CLINICAL REPORT The patient presented to our institution at the age of 28 months following treatment at an outside institution with neonatal tracheostomy for grade 3 Robin sequence. He carried the diagnosis of Treacher Collins–like variant based on a novel X-linked mutation and exhibited characteristic features of the syndrome, including midfacial hypoplasia, low-set ears, downward slanting palpebral fissures, and microretrognathia (Fig. 1). At the outside hospital, MDO was performed during early infancy in an unsuccessful attempt to decannulate the tracheostomy. The patient continued to require the tracheostomy and developed postoperative trismus. Surgical exploration and limited bilateral coronoidectomies via intraoral access resulted in minimal and temporary improvement in mandibular excursion. Upon transfer of care to our institution, the patient was unable to open his mouth and received all nutrition and hydration via gastrostomy tube. Computed tomography (CT) scan demonstrated bilateral coronoid-zygomatic fusion, and further evaluation with magnetic resonance imaging (MRI) revealed normal TMJ anatomy. The patient underwent open bilateral coronoidectomies through extended coronal and preauricular incisions (Fig.

Ms. Arnspiger is medical student, Dr. Felder is Resident, Dr. Wood is Fellow, Dr. Rogers is Assistant Professor, and Dr. Oh is Chief, Division of Plastic and Reconstructive Surgery, Children’s National Health System, Washington, DC. None of the authors has a financial interest in any of the products, devices, or drugs mentioned in this article. Submitted January 2014; Revised April 2014; Accepted April 2014. Address correspondence to: Dr. Albert K. Oh, Surgery and Pediatrics, Children’s National Health System, George Washington University, 111 Michigan Avenue, NW, Washington, DC, 20010. Email [email protected]. DOI: 10.1597/14-008 373

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FIGURE 1 A: Preoperative appearance, anterior view. Features notable for Treacher Collins–like syndrome including micrognathia with airway obstruction, midface hypoplasia, low-set ears, and downward-sloping palpebral fissures. B: Preoperative appearance, lateral view.

2). Intraoral bands of soft tissue contractures were released (Fig. 3), and bilateral TMJ manipulation was performed, resulting in approximately 2.5 cm of mandibular excursion. In the postanesthesia care unit, the patient displayed seizure-like activity of his right upper and lower extremities. A left parietal ischemic stroke in the distribution of the middle cerebral artery (Fig. 4) and a left internal carotid artery dissection at the level of the petrous portion of the temporal bone (Fig. 5) were revealed by CT and MRI.

FIGURE 2 Bilateral coronoidectomies were performed for treatment of continued pseudoankylosis.

FIGURE 3 Scar contracture bands of intraoral soft tissue were found to be contributing to the pseudoankylosis.

Arnspiger et al., COMPLICATIONS AFTER MANDIBULAR DISTRACTION

FIGURE 4 Computed tomography scan of head demonstrating left middle cerebral artery territory infarct involving a large portion of the left parietal lobe.

Paresis of the extremities improved with anticoagulation and physical therapy, and a full recovery was achieved. DISCUSSION Our patient experienced two major complications following MDO for mandibular micrognathia: TMJ ankylosis related to the distraction process and internal carotid artery dissection following correction of the first complication. True TMJ ankylosis, defined as an intracapsular bony or fibrous bridge, is a rarely reported complication of MDO (Mandell et al., 2004; Shetye et al., 2009; Master et al., 2010). It is postulated that this occurs because of a lack of cartilaginous interface between the mandibular condyle and glenoid fossa, from either syndromic abnormality or deficient reconstruction in the case of grafted bone, in addition to the stress placed on the joint as a result of the distraction vector (Stelnicki et al., 2001; Shetye et al., 2009). Inability to move the mandible can also be due to extracapsular pathology that limits mobility of the mandible, such as coronoid hyperplasia, scar contracture, or deficient musculature (pseudoankylosis). These extracapsular causes of TMJ immobility can eventually cause intracapsular pathology, particularly fibrous adhesions. The authors are not aware of a previous description of TMJ fibrous pseudoankylosis after MDO. This phenomenon has been described as a complication of radiotherapy for oral cancer (Fujioka et al., 2000), as a result of an unfavorable uterine environment leading to coronoid hyperplasia (Baraldi et al., 2010), fusion of the coronoid

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FIGURE 5 Computed tomography angiogram of the neck demonstrating contour irregularity of the left internal carotid artery before entering the petrous segment, concerning for left internal carotid artery dissection.

and zygomatic arch secondary to trauma (Lapeyrolerie et al., 1973), and secondary to congenital maxillary-mandibular synostoses (Super and Cotten, 1982). Coronoidzygomatic ankylosis is an extracapsular form of ankylosis that generally has a low incidence, with only 15 case reports identified in a recent study (Guven, 2012). In that review, 11 ¨ cases were caused by trauma, 2 were caused by infection, and 2 were subsequent to surgical interventions. The two surgical cases occurred after bilateral vertical osteotomy of mandibular rami for correction of prognathic mandible and after tumor resection from the pterygoid space with section of the mandibular body (Lindsay et al., 1966; Troyer, 1971). The second complication, internal carotid artery (ICA) dissection, can lead to stroke and death, especially in the younger population. The extracranial ICA is vulnerable to manipulation and dissection because of its mobility in the neck and fixation at the base of the skull (Patel et al., 2012). An intimal tear or rupture of the vasa vasorum leads to an intramural hematoma; this can either compress the lumen, lead to aneurysmal dilatation, or create a false lumen. Thromboembolism is the most common mechanism of stroke after ICA dissection (Patel et al., 2012), as narrowing of the lumen and intimal damage lead to thrombus formation. There are no reported occurrences of ICA dissection in association with surgical management of TMJ ankylosis. There are, however, several case reports of ICA injury after other orthognathic procedures (Lanigan, 1988; Suzuki et al., 1997; Singhal et al.,

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1998; Sigler et al., 2008; Nout et al., 2010). Injury occurs either from direct intraoperative trauma or indirect injury from forceful stretching of the ICA and cervical hyperextension with contralateral rotation of the head (Lanigan, 1988; Suzuki et al., 1997). Although rare, awareness and recognition of the potential for this complication are paramount in the treatment of this patient population. CONCLUSIONS Mandibular distraction osteogenesis is a reliable treatment for airway obstruction in neonatal Robin sequence but is not without complications. Postoperative trismus is a rare complication with multiple etiologies that are not clearly elucidated in the current literature. The cause of TMJ pseudoankylosis, whether coronoid-zygomatic fusion or soft tissue adhesions, is critical in therapeutic decision making. Correction of this postdistraction complication in our patient led to ICA dissection and stroke, a complication not previously described. REFERENCES Baraldi CE, Martins GL, Puricelli E. Pseudoankylosis of the temporomandibular joint caused by zygomatic malformation. Int J Oral Maxillofac Surg. 2010;39:729–732. Freitas RD, Tolazzi AR, Alonso N, Cruz GA, Busato L. Evaluation of molar teeth and buds in patients submitted to mandible distraction: long-term results. Plast Reconstr Surg. 2008;121:1335–1342. Fujioka M, Daian T, Murakami R, Makino K. Release of extraarticular ankylosis by coronoidectomy and insertion of a free abdominal flap: case report. J Craniofac Surg. 2000;28:369–372. Gursoy S, Hukki J, Hurmerinta K. Five year follow-up of mandibular distraction osteogenesis on the dentofacial structures of syndromic children. Orthod Craniofac Res. 2008;11:57–64. Guven O. Zygomaticocoronoid ankylosis: a rare clinical condition ¨ leading to limitation of mouth opening. J Craniofac Surg. 2012;23:829–830. Lanigan DT. Injuries to the internal carotid artery following orthognathic surgery. Int J Adult Orthodon Orthognath Surg. 1988;3:215–220. Lapeyrolerie FM, Itkin AB, Strair HC. Pseudoankylosis from fusion of coronoid process and zygomatic arch. J Oral Surg. 1973;31:788– 789. Lindsay JS, Fulcher CL, Sazima HJ, Green HG. Surgical management of ankylosis of the temporomandibular joint: report of two cases. J Oral Surg. 1966;24:264–270. Mandell DL, Yellon RF, Bradley JP, Izadi K, Gordon CB. Mandibular distraction for micrognathia and severe upper airway obstruction. Arch Otolaryngol Head Neck Surg. 2004;130:344–348. Master DL, Hanson PR, Gosain AK. Complications of mandibular distraction osteogenesis. J Craniofac Surg. 2010;21:1565–1570.

McCarthy JG, Grayson BH, Williams JK, Turk A. Distraction of the mandible: the New York University experience. In: McCarthy J, ed. Distraction of the Craniofacial Skeleton. Berlin: Springer; 1999:80– 203. McCarthy JG, Katzen T, Hopper R, Grayson BH. The first decade of mandibular distraction: lessons we have learned. Plast Reconstr Surg. 2002;110:1704–1713. McCarthy JG, Schreiber J, Karp N, Thorne CH, Grayson BH. Lengthening the human mandible by gradual distraction. Plast Reconstr Surg. 1992;89:1–8. McCarthy JG, Stelnicki EJ, Mehrara BJ, Longaker MT. Distraction osteogenesis of the craniofacial skeleton. Plast Reconstr Surg. 2001;107:1812–1827. Mofid MM, Manson PN, Robertson BC, Tufaro AP, Elias JJ, Vander Kolk CA. Craniofacial distraction osteogenesis: a review of 3278 cases. Plast Reconstr Surg. 2001;108:1103–1114. Nout E, Mathijssen IM, van der Meulen JJ, et al. Internal carotid dissection after Le Fort III distraction in Apert syndrome: a case report. J Craniofac Surg. 2010;38:529–533. Patel RR, Adam R, Maldjian C, Lincoln CM, Yuen A, Arneja A. Cervical carotid artery dissection: current review of diagnosis and treatment. Cardiol Rev. 2012;20:145–152. Shetye PR, Warren SM, Brown D, Garfinkle JS, Grayson BH, McCarthy JG. Documentation of the incidents associated with mandibular distraction: introduction of a new stratification system. Plast Reconstr Surg. 2009;123:627–634. ´ Sigler A, Gutierrez M, Harris A, Capiz M, Uriarte F. Carotid artery injury during mandibular distraction. Br J Oral Maxillofac Surg. 2008;46:419–420. Singhal A, Golomb M, Mochida G, et al. Another case of internal carotid artery dissection after mandibular osteotomy. J Oral Maxillofac Surg. 1998;56:115–116. Snyder CC, Levine GA, Swanson HM, Browne EZ Jr. Mandibular lengthening by gradual distraction: preliminary report. Plast Reconstr Surg. 1973;51:506. Stelnicki EJ, Stucki-McCormick SU, Rowe N, McCarthy JG. Remodeling of the temporomandibular joint following mandibular distraction osteogenesis in the transverse dimension. Plast Reconstr Surg. 2001;107:647–658. Super S, Cotten JS. Bilateral pseudoankylosis of the temporomandibular joint due to synostoses between the mandible and maxilla. J Oral Maxillofac Surg. 1982;40:590–592. Suzuki H, Saito E, Hashimoto K. Dissecting aneurysm of the internal carotid artery after a mandibular osteotomy. J Oral Maxillofac Surg. 1997;55:747–750. Swennen G, Schliephake H, Dempf R, Schierle H, Malevez C. Craniofacial distraction osteogenesis: a review of the literature: part 1: clinical studies. Int J Oral Maxillofac Surg. 2001;30:89–103. Troyer SH. Ankylosis of the coronoid process of the mandible to the zygomatic arch subsequent to the surgical correction of prognathism: case report. J Hosp Dent Pract. 1971;5:19–34. Uckan S, Veziroglu F, Arman A. Unexpected breakage of mandibular midline distraction device: case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006;102:e21–e25. Van Strijen PJ, Breuning KH, Becking AG, Tuinzing DB. Stability after distraction osteogenesis to lengthen the mandible: results in 50 patients. J Oral Maxillofac Surg. 2004;62:304–307.

Carotid artery dissection and stroke complicating treatment of postmandibular distraction ankylosis: a case report.

Mandibular distraction osteogenesis is an increasingly accepted treatment option for severe upper airway obstruction in grade 3 Robin sequence. Compli...
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