CRANIOMAXILLOFACIAL DEFORMITIES/COSMETIC SURGERY

Pseudoaneurysm of Internal Maxillary Artery—An Untold Complication Following Distraction Osteogenesis—A Case Report Deepak Abraham Pandyan, MDS,* Pearlcid Siroraj,y Nandakumar, MDS,z and C. D. Narayananx Pseudoaneurysm of the internal maxillary artery is very rare and only a handful of cases have been reported in the literature thus far and none after placement of a prosthetic condyle and a distraction device. This case report highlights the need for early diagnosis, appropriate steps in management, and a multidisciplinary approach in a tertiary care center in treating this life-threatening condition and proper treatment planning to prevent this condition. Ó 2014 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 72:605.e1-605.e7, 2014 In recent years, distraction osteogenesis (DO) has become an alternative to conventional surgery.1 The possible complications after DO are classified as intraoperative complications that include improper or incomplete fracturing of the osteotomized segments, complications related to the device, and bleeding.1 Intradistraction complications include infection, complications related to the device or patient, and damage to adjacent nerves.1 Postoperative complications include infection, malunion, and condylar changes.1 Pseudoaneurysm (PA) has never been mentioned in the literature as a possible complication of DO. PA, also called false aneurysm, is an extravascular hematoma after a breach in the vascular wall that freely communicates with the intravascular space.2 PAs of the maxillofacial region are usually secondary to blunt or penetrating trauma or are iatrogenic after temporomandibular joint surgeries and orthognathic surgeries.3-12 The main signs and symptoms include pain, facial asymmetry beyond the pulsatile mass, occasionally an erythematous skin overlying the swelling, neurologic deficit, and, in severe cases, thromboembolism.

The PA receives its name from the fact that it develops after rupture of the vascular endothelium, after which blood leaks out, limited only by the muscle fascia encompassing the artery. The blood flow to this extravascular compartment continues until the hematoma is large enough to counter the arterial pressure and control the hemorrhage in the affected area. The pulsating hematoma formed as a result of rupture of the endothelium develops for 1 to 8 weeks and forms a new vascular wall that may rupture and trigger severe hemorrhage or thromboembolic events. Although surgical ligation of the bleeding artery remains the gold standard, various techniques, such as covered stenting, ultrasound probe compression, ultrasound-guided thrombin injection, and embolization of the artery, also can be performed. PA of the internal maxillary artery (IMA), sphenopalatine artery, and facial artery after orthognathic surgeries, especially subcondylar osteotomies, Le Fort I osteotomies, and bilateral saggital split osteotomies, have been reported in the literature,3-13 but PA after DO has never been reported in the literature.1,14

Received from the Sri Ramachandra Medical Center, Chennai, India.

Sciences, Sri Ramachandra Medical Center, Porur, Chennai 600116,

*Assistant Professor, Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences.

Tamil Nadu, India; e-mail: [email protected] Received August 22 2013

yResident, Department of Oral and Maxillofacial Surgery, Faculty

Accepted November 11 2013

of Dental Sciences.

Ó 2014 American Association of Oral and Maxillofacial Surgeons

zProfessor, Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences.

0278-2391/13/01420-1$36.00/0

xProfessor, Department of General Surgery.

http://dx.doi.org/10.1016/j.joms.2013.11.009

Address correspondence and reprint requests to Dr Siroraj: Department of Oral and Maxillofacial Surgery, Faculty of Dental

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Report of Case

FIGURE 1. Patient when he reported to the emergency room. Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

A 26-year-old man reported to the emergency department of Sri Ramachandra Medical Center (Chennai, India) with complaints of swelling in relation to the right side of his face for the previous 10 days with several episodes of intraoral bleeding (Fig 1). A detailed history showed that the patient had been treated for an odontogenic keratocyst of the right ramus and angle of the mandible 18 days previously. The patient was treated by hemimandibulectomy and reconstruction using a prosthetic condyle and transport distraction device placement. Postoperatively, the patient had developed multiple episodes of bleeding from the intraoral surgical site. The patient underwent repeat surgery on postoperative day (POD) 18 for surgical ligation of the right facial artery. After the second procedure, the patient had 3 episodes of bleeding from the intraoral and extraoral surgical sites for more than 10 days. A suspected history of transfusion reaction after the administration of 1 U of fresh frozen plasma after the second procedure was elicited. On detailed examination, the patient was afebrile with a pulse rate of 136 beats/minute, a respiratory rate of 20 breaths/minute, systolic and diastolic blood pressures of 110 and 70 mm Hg, respectively, and no signs of toxicity. On local examination, a diffuse

FIGURE 2. Embolization of the right linguofacial trunk of the external carotid artery (arrow). Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

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FIGURE 3. Digital subtraction radiograph showing the pseudoaneurysm of the internal maxillary artery (arrow). Initially, this was considered an artifact. Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

swelling was evident on the right side of the neck extending superiorly to inferiorly from the tragus to 6 cm below the inferior border of the mandible and anteroposteriorly from the chin extending 3 cm behind the right mastoid process. The transport distraction device was evident through the chin. Sutured incisions were evident extraorally at the submandibular region and intraorally at the right retromolar area. Maculopapular rashes were evident on the shoulder and chest (suspected transfusion reaction). Baseline investigations showed the hemoglobin level to be 6 g/dL. The patient was moved to the

intensive care unit, where 2 U of packed red blood cells was transfused. Two hours after transfusion, the patient developed bleeding at the intraoral site and compression was given to control the bleeding. The blood loss was estimated at approximately 500 mL. The patient was taken for emergency embolization of the bleeding branches of the external carotid artery (ECA). Digital subtraction angiogram (DSA) depicted bleeders from the right linguofacial trunk of the ECA that had been embolized with gel foam slurry (Fig 2). A repeat DSA displayed a bilobed PA evident at the proximal first segment of the right

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FIGURE 4. Removal of the distraction device (arrow). Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

IMA adjacent to the upper knob of the metal distractor (Fig 3). Subsequently, the patient was taken for surgical ligation of the right maxillary artery under general anesthesia. The patient was placed in a supine position and general anesthesia was administered through endotracheal intubation. The distraction device was removed (Fig 4). The bleeding site was identified by performing a lip-split incision extending to the existing submandibular skin incision. The right infratemporal and glenoid fossae were explored through the incision (Fig 5). The IMA was ligated at the

bifurcation from the right ECA with the help of 30 Prolene suture. A gauze pack was placed at the dead space and a free end of the gauze pack was brought out through the submandibular incision (Fig 6). Layer-by-layer closure was performed. The patient was shifted to the intensive care unit without extubation for observation (Fig 7). The patient was extubated on POD 2 and a Ryle tube feeding was started on POD 2. The pack placed in the glenoid fossa was removed on POD 2 and the surgical site was checked for active bleeding. The patient was closely monitored and oral feeds were

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FIGURE 5. Lip-split incision (black arrow) and ligation of the right internal maxillary artery (yellow arrow). Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

started on POD 5. The patient’s vitals were stable. Because he tolerated oral feeds well, the patient was discharged on POD 5 and is under regular monthly follow-up (Fig 8).

Discussion The IMA is the terminal and largest branch of the ECA and is divided into 3 parts, namely the proximal mandibular, middle pterygoid, and terminal pterygopalatine segments.15 The pterygopalatine segment is commonly associated with the formation of a PA,15,16 whereas the proximal mandibular part was involved in the present case. Various complications encountered include oronasal hemorrhage after rupture of the PA,

compression of the adjacent nerves or artery, and thromboembolic events.15 PA often presents as an expanding pulsatile mass. The common etiology of PA of the IMA includes blunt and penetrating traumas, facial fractures, and iatrogenic causes, such as orthognathic surgery, neck dissection after head and neck cancer, surgical removal of impacted third molars, and radiotherapy.3-13,16,17 Other rare causes include infections, atherosclerosis, cystic medial necrosis, and fibromuscular dysplasia, which can lead to PA of the ECA rather than to the branches of the ECA.16 The etiology in this case was the distraction device at the glenoid fossa, which had traumatized the IMA. PA is not listed as a possible complication of DO in

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FIGURE 6. Gauze pack in the dead space (arrow). Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

the literature,1,14 but the potential of a distraction device causing this condition is evident. Therefore, care must be taken when placing a distraction device to avoid injury to the adjacent vital structures. Catheter angiography remains the gold standard in diagnosing PA of the IMA. Other diagnosing aids, such as color Doppler and computed tomography, are useful in diagnosing PAs larger than 1 cm.16,18 Any delay in diagnosing and managing this condition leads to life-threatening complications.3 The treatment protocol for PA starts with packing and compression to arrest oral and nasal bleeding. Transfusion of blood is usually required to maintain vital signs. Surgical liga-

tion remains the treatment of choice in cases in which selective embolization using microcatheter systems have failed. The advantages of selective embolization over surgical ligation are that it is less invasive and can be performed under local anesthesia. It can be performed with diagnostic angiography, thus preserving other branches of the main trunk with improved access. The main associated drawbacks are technique sensitivity, cost, and artifacts on the DSA, which can lead to misinterpretations as encountered in the present case. PAs of the ECA are rare but life-threatening and require immediate intervention. McCollum et al19 in

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their study of 8,000 PAs over a period of 21 years reported only 21 PAs of the ECA, of which 19 cases were due to iatrogenic causes. In most cases in the literature, PA is iatrogenic so careful treatment planning and proper knowledge about the anatomy of the adjacent vital structures, proper visualization, and careful instrumentation are required to prevent this condition.

References

FIGURE 7. Closure of the surgical wound. Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

FIGURE 8. One week postoperatively. Pandyan et al. Pseudoaneurysm of Internal Maxillary Artery. J Oral Maxillofac Surg 2014.

1. van Strijen PJ, Breuning KH, Becking AG, et al: Complications in bilateral mandibular distraction osteogenesis using internal devices. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 96: 392, 2003 2. Kumar, Abbas, Fausto et al (eds): Robbins Textbook of Basic Pathology (ed 8). Philadelphia, PA, Saunders/Elsevier 3. Gerbino G, Rocia F, Grosso M, et al: Pseudoaneurysm of the internal maxillary artery and Frey’s syndrome after blunt facial trauma. J Oral Maxillofac Surg 55:1484, 1997 4. Clark R, Lew D, Giyanani VL, et al: False aneurysm complicating orthognathic surgery. J Oral Maxillofac Surg 45:57, 1987 5. de Lucas EM, Gutierrez A, Mandly AG, et al: Life-threatening pseudoaneurysm of the facial artery after dental extraction: successful treatment with emergent endovascular embolization. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 106:129, 2008 6. Pappa H, Richardson D, Niven S: False aneurysm of the facial artery as complication of sagittal split osteotomy. J Craniomaxillofac Surg 36:180, 2008 7. Cohen MA: False (traumatic) aneurysm of the facial artery caused by a foreign body. Int J Oral Maxillofac Surg 15:336, 1986 8. Dediol E, Manojlovic S, Biocic J, et al: Facial artery pseudoaneurysm without evidence of trauma. Int J Oral Maxillofac Surg 40: 988, 2011 9. Bays RA, Bouloux GF: Complications of orthognathic surgery. Oral Maxillofac Surg Clin North Am 15:229, 2003 10. Bradley JP, Elahi M, Kawamoto HK: Delayed presentation of pseudoaneurysm after Le Fort I osteotomy. J Craniofac Surg 13:746, 2002 11. Lanigan DT, Hey JH, West RA: Major vascular complications of orthognathic surgery: False aneurysms and arteriovenous fistulas following orthognathic surgery. J Oral Maxillofac Surg 49: 571, 1991 12. Hemmig SB, Johnson RS, Ferraro N: Management of a ruptured pseudoaneurysm of the sphenopalatine artery following a Le Fort I osteotomy. J Oral Maxillofac Surg 45:533, 1987 13. Elton VJF, Turnbull IW, Foster ME: An overview of the management of pseudo aneurysm of the maxillary artery: A report of a case following mandibular subcondylar osteotomy. J Craniomaxillofac Surg 35:52, 2007 14. Nørholt SE, Jensen J, Schou S, et al: Complications after mandibular distraction osteogenesis: A retrospective study of 131 patients. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 111:420, 2011 15. Osborn AG: The external carotid artery, in Osborn AG, (ed). Diagnostic Cerebral Angiography (ed 2). Philadelphia. PA, Lippincott Williams & Wilkins, 1999, pp 49–55 16. Luo C-B, Teng MM-H, Chang F-C, et al: Role of CT and endovascular embolization in managing pseudoaneurysms of the internal maxillary artery. J Chin Med Assoc 69:310, 2006 17. Peoples JR III, Herbosa EG, Dion J: Management of internal maxillary artery hemorrhage from temporomandibular joint surgery via selective embolization. J Oral Maxillofac Surg 46:1005, 1988 18. Rogers SN, Patel M, Beirne JC, et al: Traumatic aneurysm of the maxillary artery: The role of interventional radiology. Int J Oral Maxillofac Surg 24:336, 1995 19. McCollum CH, Wheeler WG, Noon GP, et al: Aneurysm of the extracranial carotid artery. Twenty-one years’ experience. Am J Surg 137:196, 1979

Pseudoaneurysm of internal maxillary artery--an untold complication following distraction osteogenesis--a case report.

Pseudoaneurysm of the internal maxillary artery is very rare and only a handful of cases have been reported in the literature thus far and none after ...
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