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Internal Carotid Artery Pseudoaneurysm After Le Fort I Osteotomy: Report of a Case and Its Management
J Oral Maxillofac Surg 69:e242-e245, 2011
Internal Carotid Artery Pseudoaneurysm After Le Fort I Osteotomy: Report of a Case and Its Management Deepak G. Krishnan, DDS,* Dale L. Alto, DDS,† Theron C. Waisath, DMD,‡ Andrew W. Grande, MD,§ Usman Khan, MD,储 and Todd Abruzzo, MD¶ Case Report
In the treatment of maxillofacial deformities, the Le Fort I osteotomy is a widely used and versatile procedure. However, complications associated with this procedure are well described and attest to its inherent dangers. The most serious and life-threatening complications are vascular injuries. As reported in the literature, complications primarily involve injury to branches of the external carotid artery, especially the internal maxillary artery, and numerous accounts of intraoperative hemorrhages and management. Accounts of traumatic aneurysms located on branches of the external carotid also exist, yet appear to be rare occurrences. Most of these aneurysms occur in a delayed manner and are primarily associated with projectile injuries or blunt trauma. This case report describes a unique complication, a vascular injury to the internal carotid artery (ICA) in a patient who underwent a Le Fort I osteotomy as part of a procedure to establish a proper maxillomandibular relationship.
A 33-year-old man was referred to the University of Cincinnati, Oral and Maxillofacial Surgery Service for evaluation of a Class III skeletal and dental malocclusion and multiple carious teeth. The patient’s medical and surgical history was unremarkable except for the repair of a bilateral cleft lip that was performed at an unknown time. On clinical examination, the patient exhibited an anterior-posterior maxillomandibular discrepancy of 12 mm. Cephalometric analysis confirmed the clinical findings of maxillary hypoplasia. The patient’s mandible appeared to be in good position both clinically and radiographically. A preoperative computed tomographic (CT) scan showed an untreated unilateral alveolar cleft. A 2-phase treatment plan was developed based on the clinical and radiographic findings as well as study models and a stereolithographic model. In phase I, a Le Fort I osteotomy is performed with placement of internal maxillary distractors along with a bone graft to the alveolar cleft. After distraction of the patient’s maxilla to establish a proper maxillomandibular relationship, phase II would include removal of the necessary teeth followed by rehabilitation that would include dental implants and complete dentures. During phase I, the patient successfully underwent a Le Fort I osteotomy with placement of internal maxillary distractors and an alveolar cleft bone graft in November 2008. On postoperative day 1, the patient reported a persistent left upper extremity weakness and a sore throat. Although these symptoms were clinically evaluated by the neurology service who initially did not suspect any central neurologic problem, brain magnetic resonance imaging would later confirm an ischemic stroke in the territory of the right middle cerebral artery. During the patient’s hospital stay, he continued to note a worsening sore throat and mild dysphagia; however, no underlying cause was discovered. Because the patient had no respiratory distress and continued to maintain adequate oral intake, he was discharged on postoperative day 4. Four days after discharge, the patient returned to the outpatient clinic for routine evaluation of the
*Assistant Professor of Surgery and Residency Program Director, Division of Oral Maxillofacial Surgery, Department of Surgery, University of Cincinnati, Cincinnati, OH. †Former Chief Resident, Division of Oral Maxillofacial Surgery, Department of Surgery, University of Cincinnati, Cincinnati, OH; Currently, Private Practice, Colorado Springs, CO. ‡Former Chief Resident, Division of Oral Maxillofacial Surgery, Department of Surgery University of Cincinnati, Cincinnati, OH; Currently, Private Practice, Champaign, IL. §Cerebrovascular/Endovascular Fellow, Department of Neurosurgery, University of Cincinnati, Cincinnati, OH. 储Cerebrovascular/Endovascular Fellow, Department of Neurosurgery, University of Cincinnati, Cincinnati, OH. ¶Associate Professor of Neurosurgery, Radiology and Biomedical Engineering, Mayfield Clinic and Department of Neurosurgery, University of Cincinnati, Cincinnati, OH. Address correspondence and reprint requests to Dr Krishnan: University of Cincinnati, Division of Oral Maxillofacial Surgery, PO Box 670558, Cincinnati, OH 45267-0558; e-mail: [email protected] © 2011 American Association of Oral and Maxillofacial Surgeons. Published
by Elsevier Inc. All rights reserved. 0278-2391/11/6906-0081$36.00/0 doi:10.1016/j.joms.2011.01.035
e242
e243
KRISHNAN ET AL
FIGURE 1. CT angiogram depicting a right carotid pseudoaneurysm after a Le Fort I procedure. (A) A coronal section of a CT angiogram after a Le Fort I procedure (white arrow). A large circumscribed aneurysm is seen (arrowhead) medial to the pterygoid plate. (B) Sagittal section of a CT angiogram after a Le Fort I procedure (white arrow). A large circumscribed aneurysm arises off the ICA (arrowhead) adjacent to where the ICA enters the skull base. Krishnan et al. Internal Carotid Artery Pseudoaneurysm. J Oral Maxillofac Surg 2011.
distraction process. With the continued complaint of progressive dysphagia that was now associated with respiratory distress, hoarseness, intractable cough, inability to swallow, tachycardia, and lethargy, the patient was readmitted for intravenous fluid hydration and evaluation of his dysphagia. A modified bariumswallow study and a nasopharyngoscopy showed an arytenoid cartilage dislocation. During the course of this hospitalization, the patient developed severe right facial pain and was noted to have right-sided ptosis and meiosis. A contrast-enhanced CT scan showed a pseudoaneurysm of the right ICA measuring 3.5 cm in diameter (Figs 1, 2). Diagnostic cerebral angiography and test balloon occlusion of the right
ICA were performed. The angiogram clearly showed a complex pseudoaneurysm with 2 communicating chambers measuring 11 ⫻ 10 ⫻ 8 mm and 30 ⫻ 30 ⫻ 27 mm, respectively, and extending to the level of the odontoid process. Although the patient passed the test occlusion (using both clinical and scintigraphic criteria), we elected to attempt endovascular vessel reconstruction rather than perform therapeutic carotid occlusion. We did not consider an open surgical approach because of the anticipated difficulty in obtaining distal control over the ICA. Due to worsening respiratory distress and concerns that acute thrombosis of the pseudoaneurysm would further aggravate the patient’s airway compression, tracheostomy and
e244
INTERNAL CAROTID ARTERY PSEUDOANEURYSM
FIGURE 2. Digital subtraction angiograms depicting a pseudoaneurysm arising from the internal carotid artery. Digital subtraction angiograms demonstrating the pseudoaneurysm arising from the ICA before, during, and after treatment. (A) Before treatment the aneurysm filled briskly from the ICA. Contrast is seen mixing with unopacified blood within the aneurysm. (B) After microcoil embolization no further filling of the aneurysm is observed. Krishnan et al. Internal Carotid Artery Pseudoaneurysm. J Oral Maxillofac Surg 2011.
percutaneous gastrostomy were performed before initiating the endovascular therapy. Endovascular therapy was performed with the intent to exclude the pseudoaneurysm by placement of overlapping telescoping stents across the pseudoaneurysmal ostium. After deployment of 2 coaxial, overlapping Precise® stents (Johnson and Johnson Healthcare Systems, Piscataway, NJ) (8 ⫻ 40 mm), there was persistent flow into the pseudoaneurysm; placement of a third coaxial Precise stent was attempted but technically unsuccessful. After the pseudoaneurysm was accessed across the coaxial stent assembly with a microcatheter, coil embolization of the pseudoaneu-
rysm sac was performed (Fig 3). The patient tolerated the procedure well, was observed postoperatively in the intensive care unit, was transferred to the floor the following day, and was discharged to a long-term rehabilitation center 11 days later. At 6-week follow-up examination, his tracheotomy was decannulated and the gastrostomy tube was removed. His wounds had healed; he had no symptoms of cough or hoarseness, and had returned to work and was living independently without any significant disability. The 6-month follow-up angiography showed complete occlusion of the pseudoaneurysm with no evidence of flow-significant intimal hyperplasia within the stented ICA.
e245
KRISHNAN ET AL
FIGURE 3. Postoperative panograph depicting postmicrocoil embolization of right ICA pseudoaneurysm. Krishnan et al. Internal Carotid Artery Pseudoaneurysm. J Oral Maxillofac Surg 2011.
Discussion Traumatic pseudoaneurysms located near the skull base are hazardous to approach and challenging to repair.1 In the past, surgical treatment of ICA pseudoaneurysms has been the preferred approach2,3; however, the close proximity to the skull base makes repair difficult. The development of endovascular techniques now provides a feasible alternative to direct surgical repair for vascular injuries of the ICA near the skull base.1,4 Our patient underwent a successful endovascular treatment of a unique complication, specifically an ICA pseudoaneurysm after a Le Fort I osteotomy for severe maxilla-mandibular malocclusion. A pseudoaneurysm is a transmural breech of arterial wall integrity that extends across all layers of the arterial wall and is contained only by a fibrin capsule. In most cases, the pressure from the perivascular hematoma counterbalances the arterial pressure, thus stabilizing the hemorrhage. After several weeks, liquefaction of the hematoma begins. During this process, the pseudoaneurysm progressively enlarges which can lead to compression of adjacent nerves, release of thromboemboli, and in some cases rupture with life-threatening hemorrhage.5 Pseudoaneurysms are uncommon in the maxillofacial region and usually result from blunt or penetrating injury, or surgical trauma.6 In a series of 8,500 aneurysms, 21 of which involved the extracranial carotid system, McCollum et al reported that only 2 were a result of traumatic injury.7 Studies show that the most common location for iatrogenic pseudoaneurysms after maxillary orthognathic surgery is the internal maxillary artery.8 Other locations, such as the lingual, superficial temporal, and facial arteries, have also been reported.9-11
When suspicion of pseudoaneurysm is raised by clinical findings, a diagnosis is then confirmed based on clinical and radiographic findings. Angiography and contrast-enhanced CT scan are the preferred radiographic modalities used for diagnosis. In conclusion, the orthognathic surgeon should be aware of the possibility of occult vascular injury after orthognathic surgery.
References 1. Redekop G, Marotta T, Weill A: Treatment of traumatic aneurysms and arteriovenous fistulas of the skull base by using endovascular stents. J Neurosurg 95:412, 2001 2. Ramadan F, Rutledge R, Oller D, et al: Carotid artery trauma: A review of contemporary trauma center experiences. J Vas Surg 21:46, 1995 3. D’Alise MD, Vardiman AB, Kopitnik TA Jr, et al: External carotid to middle cerebral bypass in the treatment of complex internal carotid injury. J Trauma 40:452, 1996 4. Salgarelli A, Morana G, Beltramello A: Pseudoaneurysm of the lingual artery: A case report. J Oral Maxillofac Surg 55:860, 1997 5. Silva AC, O’Ryan F, Beckley ML, et al: Pseudoaneurysm of a branch of the maxillary artery following mandibular ramus osteotomy: Case report and review of the literature. J Oral Maxillofac Surg 65:1807, 2007 6. Pappa H, Richardson D, Niven S: False aneurysm of the facial artery as a complication of sagittal split osteotomy. J CranioMaxillofac Surg 36:180, 2008 7. McCollum C, II, Wheeler WG, Noon GP, et al: Aneurysms of the extracranial carotid artery, twenty one years’ experience. Am J Surg 137:196, 1979 8. Rogers SN: Traumatic aneurysm of the maxillary artery: The role of interventional radiology. A report of two cases. Int J Oral Maxillofac Surg 24:336, 1995 9. Conner WC, Rodrich RJ, Pollock RA: Traumatic aneurysms of the face and temple: A patient report and literature review, 1664-1998. Ann Plast Surg 41:321, 1998 10. Van den Akker HP, van der Lijn F: A false aneurysm of the facial artery as a complication of circumferential wiring. J Oral Surg 37:514, 1974 11. Fleischer AS, Guthkelch AN: Management of high cervicalintracranial internal carotid artery traumatic aneurysms. J Trauma 27:330, 1987
Internal Carotid Artery Pseudoaneurysm After Le Fort I Osteotomy: Report of a Case and Its Management Deepak G. Krishnan, DDS,* Dale L. Alto, DDS,† Theron C. Waisath, DMD,‡ Andrew W. Grande, MD,§ Usman Khan, MD,储 and Todd Abruzzo, MD¶ Case Report
In the treatment of maxillofacial deformities, the Le Fort I osteotomy is a widely used and versatile procedure. However, complications associated with this procedure are well described and attest to its inherent dangers. The most serious and life-threatening complications are vascular injuries. As reported in the literature, complications primarily involve injury to branches of the external carotid artery, especially the internal maxillary artery, and numerous accounts of intraoperative hemorrhages and management. Accounts of traumatic aneurysms located on branches of the external carotid also exist, yet appear to be rare occurrences. Most of these aneurysms occur in a delayed manner and are primarily associated with projectile injuries or blunt trauma. This case report describes a unique complication, a vascular injury to the internal carotid artery (ICA) in a patient who underwent a Le Fort I osteotomy as part of a procedure to establish a proper maxillomandibular relationship.
A 33-year-old man was referred to the University of Cincinnati, Oral and Maxillofacial Surgery Service for evaluation of a Class III skeletal and dental malocclusion and multiple carious teeth. The patient’s medical and surgical history was unremarkable except for the repair of a bilateral cleft lip that was performed at an unknown time. On clinical examination, the patient exhibited an anterior-posterior maxillomandibular discrepancy of 12 mm. Cephalometric analysis confirmed the clinical findings of maxillary hypoplasia. The patient’s mandible appeared to be in good position both clinically and radiographically. A preoperative computed tomographic (CT) scan showed an untreated unilateral alveolar cleft. A 2-phase treatment plan was developed based on the clinical and radiographic findings as well as study models and a stereolithographic model. In phase I, a Le Fort I osteotomy is performed with placement of internal maxillary distractors along with a bone graft to the alveolar cleft. After distraction of the patient’s maxilla to establish a proper maxillomandibular relationship, phase II would include removal of the necessary teeth followed by rehabilitation that would include dental implants and complete dentures. During phase I, the patient successfully underwent a Le Fort I osteotomy with placement of internal maxillary distractors and an alveolar cleft bone graft in November 2008. On postoperative day 1, the patient reported a persistent left upper extremity weakness and a sore throat. Although these symptoms were clinically evaluated by the neurology service who initially did not suspect any central neurologic problem, brain magnetic resonance imaging would later confirm an ischemic stroke in the territory of the right middle cerebral artery. During the patient’s hospital stay, he continued to note a worsening sore throat and mild dysphagia; however, no underlying cause was discovered. Because the patient had no respiratory distress and continued to maintain adequate oral intake, he was discharged on postoperative day 4. Four days after discharge, the patient returned to the outpatient clinic for routine evaluation of the
*Assistant Professor of Surgery and Residency Program Director, Division of Oral Maxillofacial Surgery, Department of Surgery, University of Cincinnati, Cincinnati, OH. †Former Chief Resident, Division of Oral Maxillofacial Surgery, Department of Surgery, University of Cincinnati, Cincinnati, OH; Currently, Private Practice, Colorado Springs, CO. ‡Former Chief Resident, Division of Oral Maxillofacial Surgery, Department of Surgery University of Cincinnati, Cincinnati, OH; Currently, Private Practice, Champaign, IL. §Cerebrovascular/Endovascular Fellow, Department of Neurosurgery, University of Cincinnati, Cincinnati, OH. 储Cerebrovascular/Endovascular Fellow, Department of Neurosurgery, University of Cincinnati, Cincinnati, OH. ¶Associate Professor of Neurosurgery, Radiology and Biomedical Engineering, Mayfield Clinic and Department of Neurosurgery, University of Cincinnati, Cincinnati, OH. Address correspondence and reprint requests to Dr Krishnan: University of Cincinnati, Division of Oral Maxillofacial Surgery, PO Box 670558, Cincinnati, OH 45267-0558; e-mail: [email protected] © 2011 American Association of Oral and Maxillofacial Surgeons. Published
by Elsevier Inc. All rights reserved. 0278-2391/11/6906-0081$36.00/0 doi:10.1016/j.joms.2011.01.035
e242
e243
KRISHNAN ET AL
FIGURE 1. CT angiogram depicting a right carotid pseudoaneurysm after a Le Fort I procedure. (A) A coronal section of a CT angiogram after a Le Fort I procedure (white arrow). A large circumscribed aneurysm is seen (arrowhead) medial to the pterygoid plate. (B) Sagittal section of a CT angiogram after a Le Fort I procedure (white arrow). A large circumscribed aneurysm arises off the ICA (arrowhead) adjacent to where the ICA enters the skull base. Krishnan et al. Internal Carotid Artery Pseudoaneurysm. J Oral Maxillofac Surg 2011.
distraction process. With the continued complaint of progressive dysphagia that was now associated with respiratory distress, hoarseness, intractable cough, inability to swallow, tachycardia, and lethargy, the patient was readmitted for intravenous fluid hydration and evaluation of his dysphagia. A modified bariumswallow study and a nasopharyngoscopy showed an arytenoid cartilage dislocation. During the course of this hospitalization, the patient developed severe right facial pain and was noted to have right-sided ptosis and meiosis. A contrast-enhanced CT scan showed a pseudoaneurysm of the right ICA measuring 3.5 cm in diameter (Figs 1, 2). Diagnostic cerebral angiography and test balloon occlusion of the right
ICA were performed. The angiogram clearly showed a complex pseudoaneurysm with 2 communicating chambers measuring 11 ⫻ 10 ⫻ 8 mm and 30 ⫻ 30 ⫻ 27 mm, respectively, and extending to the level of the odontoid process. Although the patient passed the test occlusion (using both clinical and scintigraphic criteria), we elected to attempt endovascular vessel reconstruction rather than perform therapeutic carotid occlusion. We did not consider an open surgical approach because of the anticipated difficulty in obtaining distal control over the ICA. Due to worsening respiratory distress and concerns that acute thrombosis of the pseudoaneurysm would further aggravate the patient’s airway compression, tracheostomy and
e244
INTERNAL CAROTID ARTERY PSEUDOANEURYSM
FIGURE 2. Digital subtraction angiograms depicting a pseudoaneurysm arising from the internal carotid artery. Digital subtraction angiograms demonstrating the pseudoaneurysm arising from the ICA before, during, and after treatment. (A) Before treatment the aneurysm filled briskly from the ICA. Contrast is seen mixing with unopacified blood within the aneurysm. (B) After microcoil embolization no further filling of the aneurysm is observed. Krishnan et al. Internal Carotid Artery Pseudoaneurysm. J Oral Maxillofac Surg 2011.
percutaneous gastrostomy were performed before initiating the endovascular therapy. Endovascular therapy was performed with the intent to exclude the pseudoaneurysm by placement of overlapping telescoping stents across the pseudoaneurysmal ostium. After deployment of 2 coaxial, overlapping Precise® stents (Johnson and Johnson Healthcare Systems, Piscataway, NJ) (8 ⫻ 40 mm), there was persistent flow into the pseudoaneurysm; placement of a third coaxial Precise stent was attempted but technically unsuccessful. After the pseudoaneurysm was accessed across the coaxial stent assembly with a microcatheter, coil embolization of the pseudoaneu-
rysm sac was performed (Fig 3). The patient tolerated the procedure well, was observed postoperatively in the intensive care unit, was transferred to the floor the following day, and was discharged to a long-term rehabilitation center 11 days later. At 6-week follow-up examination, his tracheotomy was decannulated and the gastrostomy tube was removed. His wounds had healed; he had no symptoms of cough or hoarseness, and had returned to work and was living independently without any significant disability. The 6-month follow-up angiography showed complete occlusion of the pseudoaneurysm with no evidence of flow-significant intimal hyperplasia within the stented ICA.
e245
KRISHNAN ET AL
FIGURE 3. Postoperative panograph depicting postmicrocoil embolization of right ICA pseudoaneurysm. Krishnan et al. Internal Carotid Artery Pseudoaneurysm. J Oral Maxillofac Surg 2011.
Discussion Traumatic pseudoaneurysms located near the skull base are hazardous to approach and challenging to repair.1 In the past, surgical treatment of ICA pseudoaneurysms has been the preferred approach2,3; however, the close proximity to the skull base makes repair difficult. The development of endovascular techniques now provides a feasible alternative to direct surgical repair for vascular injuries of the ICA near the skull base.1,4 Our patient underwent a successful endovascular treatment of a unique complication, specifically an ICA pseudoaneurysm after a Le Fort I osteotomy for severe maxilla-mandibular malocclusion. A pseudoaneurysm is a transmural breech of arterial wall integrity that extends across all layers of the arterial wall and is contained only by a fibrin capsule. In most cases, the pressure from the perivascular hematoma counterbalances the arterial pressure, thus stabilizing the hemorrhage. After several weeks, liquefaction of the hematoma begins. During this process, the pseudoaneurysm progressively enlarges which can lead to compression of adjacent nerves, release of thromboemboli, and in some cases rupture with life-threatening hemorrhage.5 Pseudoaneurysms are uncommon in the maxillofacial region and usually result from blunt or penetrating injury, or surgical trauma.6 In a series of 8,500 aneurysms, 21 of which involved the extracranial carotid system, McCollum et al reported that only 2 were a result of traumatic injury.7 Studies show that the most common location for iatrogenic pseudoaneurysms after maxillary orthognathic surgery is the internal maxillary artery.8 Other locations, such as the lingual, superficial temporal, and facial arteries, have also been reported.9-11
When suspicion of pseudoaneurysm is raised by clinical findings, a diagnosis is then confirmed based on clinical and radiographic findings. Angiography and contrast-enhanced CT scan are the preferred radiographic modalities used for diagnosis. In conclusion, the orthognathic surgeon should be aware of the possibility of occult vascular injury after orthognathic surgery.
References 1. Redekop G, Marotta T, Weill A: Treatment of traumatic aneurysms and arteriovenous fistulas of the skull base by using endovascular stents. J Neurosurg 95:412, 2001 2. Ramadan F, Rutledge R, Oller D, et al: Carotid artery trauma: A review of contemporary trauma center experiences. J Vas Surg 21:46, 1995 3. D’Alise MD, Vardiman AB, Kopitnik TA Jr, et al: External carotid to middle cerebral bypass in the treatment of complex internal carotid injury. J Trauma 40:452, 1996 4. Salgarelli A, Morana G, Beltramello A: Pseudoaneurysm of the lingual artery: A case report. J Oral Maxillofac Surg 55:860, 1997 5. Silva AC, O’Ryan F, Beckley ML, et al: Pseudoaneurysm of a branch of the maxillary artery following mandibular ramus osteotomy: Case report and review of the literature. J Oral Maxillofac Surg 65:1807, 2007 6. Pappa H, Richardson D, Niven S: False aneurysm of the facial artery as a complication of sagittal split osteotomy. J CranioMaxillofac Surg 36:180, 2008 7. McCollum C, II, Wheeler WG, Noon GP, et al: Aneurysms of the extracranial carotid artery, twenty one years’ experience. Am J Surg 137:196, 1979 8. Rogers SN: Traumatic aneurysm of the maxillary artery: The role of interventional radiology. A report of two cases. Int J Oral Maxillofac Surg 24:336, 1995 9. Conner WC, Rodrich RJ, Pollock RA: Traumatic aneurysms of the face and temple: A patient report and literature review, 1664-1998. Ann Plast Surg 41:321, 1998 10. Van den Akker HP, van der Lijn F: A false aneurysm of the facial artery as a complication of circumferential wiring. J Oral Surg 37:514, 1974 11. Fleischer AS, Guthkelch AN: Management of high cervicalintracranial internal carotid artery traumatic aneurysms. J Trauma 27:330, 1987