J Oral Maxillofac 49571-577.
Surg
1991
Major Vascular Complications Orthogna thic Surgery:
of
False Aneurysms and Arteriovenous Fistulas Following Orthogna thic Surgery DENNIS T. LANIGAN, DMD, MD,* JULIANA H. HEY, DDS, MD,t AND ROGER A. WEST, DMDS False aneurysms and arteriovenous fistulas are rare complications of orthognathic surgery. The vessel most commonly involved with false aneurysms following mandibular surgery is the internal maxillary artery, and this vessel, especially the sphenopalatine branch, may also be involved following maxillary surgery. An unusual factor in the presentation of false aneurysms following Le Fort I osteotomies is an initial episode of epistaxis occurring greater than 2 weeks postoperatively. Arteriovenous fistulas following orthognathic surgery are more apt to involve large vessels, especially the internal carotid artery. Embolization procedures are the treatment of choice for false aneurysms and arteriovenous fistulas in the maxillofacial region following orthognathic surgery.
aneurysm usually results from a tangential, incomplete tear of the arterial vessel wall so that the continuity of blood flow through the lacerated artery is generally maintained.13 Hemorrhage occurs into the adjacent soft tissues surrounding the artery until hematoma formation compresses the area and tamponades the bleeding. The hemorrhage continues until pressure in the periarterial zone equals the mean arterial pressure.’ The hematoma subsequently organizes at the periphery, with the perivascular connective tissue creating a sac around which an endothelial lining forms to produce a pseudointima that remains in continuity with the arterial lumen.**14 With liquefaction of the hematoma, a communication develops between the artery and the aneurysmal sac resulting in a pulsating mass. The arterial pressure leads to gradual expansion of the false aneurysm, which can either rupture or continue to progressively enlarge.‘,‘3*‘9 An incomplete laceration of an artery in conjunction with a concomitant injury to its accompanying vein can lead to an abnormal communication between them, resulting in an A-V tistula. The incomplete disruption of the arterial wall results in hemorrhage and hematoma formation that eventually tamponades the bleeding. The mechanism for the development of a traumatic A-V fistula has been postulated to be the organization of a hematoma
False aneurysms (pseudoaneursysms) and arteriovenous (A-V) fistulas have only rarely been reported following orthognathic surgery,le8 facial trauma,““’ or other surgical procedures in the head and neckmaxillofacial region.‘2-24 The small size of most blood vessels in the facial region makes their partial transection unlikely, and this is the principal reason for the rarity of false aneurysms or A-V fistulas in this locationg3” Penetrating or blunt trauma can lead to arterial damage. A traumatic intimal laceration can act as the site for occlusion of the vessel or for the origin of a false aneurysm. A more severe disruption of the arterial wall could lead to hemorrhage or the formation of an A-V fistula.4*25 A false
* Professor and Head, Division of Oral and Maxillofacial Surgery, University of Saskatchewan. t Formerly Clinical Fellow, Division of Oral and Maxillofacial Surgery, University of Saskatchewan; currently, Clinical Instructor, College of Dentistry, University of Saskatchewan. $ Director, Northwest Center for Corrective Jaw Surgery, Seattle. Address correspondence and reprint requests to Dr Lanigan: Division of Oral and Maxillofacial Surgery, College of Dentistry, University of Saskatchewan, Saskatoon, Saskatchewan, S7N OWOCanada. 0 1991 geons 0278-2391
American
Association
of Oral
and Maxillofacial
Sur-
I91 /4906-0004$3.00/O
571
572
ORTHOGNATHIC
arising from damage to the vaso vasorum of the artery. 24 Endothelial bud proliferation into the organizing hematoma can lead to multiple endothelial lined channels forming between the damaged arterial and venous systems rather than just a single communication.” A listula may be established immediately, or it may not become apparent for days or weeks. A shunting of blood from the arterial to the venous system results in a bypassing of the capillary system resistance and peripheral blood flow is decreased in the involved artery. The decreased vascular resistance at the site of the flstula leads to blood flow tending to preferentially enter the A-V fistula and to the progressive development of an extensive collateral circulation around the fistula. The increased blood flow to the area produces a dilation of the feeding arteries, while increased pressure in the draining veins results in them becoming dilated and thickened.15 An A-V fistula can produce a continuous vibratory thrill and a bruit, which is intensified during systole. There is wide transmission of a “machinery-like” murmur.t2*t3 With false aneurysms, depending on the amount of thrombus within the aneurysmal sac, the mass could be pulsatile and a bruit may be present. l9 The bruit is generally heard in systole and remains localized. I2 With A-V Iistulas there can be an absence or a more localized degree of swelling than is seen with the continuously expanding sac of a false aneu12,13 The abundant collateral circulation that rysm. develops with an A-V Iistula is in contrast to the poor collateral circulation found with the false aneurysm. I2 The dilation and pulsation of local veins that can be seen in an A-V listula are not seen with false aneurysms. Untreated false aneurysms may progressively enlarge until they rupture, leading to significant hemorrhage. l3 Enlargement without rupture can lead to facial asymmetry if the lesion is located near the surface of the skin. Pressure on nerves from the enlargement of a pseudoaneurysm also can result in pain or paresis.15 On the other hand, a localized A-V fistula can be relatively indolent, with no progressive enlargement or tendency to rupture. l2 On radiographic examination, false aneurysms are characterized by residual contrast in the lesion long after the arterial phase of angiography is completed.’ False aneurysms can be caused by blunt’2,13*17or, more commonly, by penetrating trauma. Gunshot wounds or knife injuries account for the majority of cases9*13 although vessels can also be lacerated by facial fractures’4*‘8’19’21 or the placement of a circumandibular wire. 2o The superficial temporal and facial arteries are the vessels in the head and neck region most commonly involved in traumatic false aneurysms. l1 The superficial temporal artery is es-
SURGERY-FALSE
ANEURYSMS
AND A-V FISTULAS
pecially vulnerable to injury due to its long and superficial course.9 The relatively small size, and deep, well-protected, location of the maxillary artery undoubtedly contribute to the rare occurrence of posttraumatic pseudoaneurysms in this vessel. In the posttraumatic cases that have been reported, two cases were related to penetrating trauma,‘.” one to compound fractures of the mandibular ramus and condylar neck,” and one to a Le Fort II and mandibular fracture.” Miller et a121reported a case of a large false aneurysm from the sphenopalatine branch of the maxillary artery in a young man who developed severe epistaxis following a Le Fort III fracture of the maxilla. Four cases of false aneurysm following orthognathic surgery have been reported to date. Clark et al’ reported the case of a 15year-old boy who underwent bilateral mandibular vertical subcondylar osteotomies for the correction of mandibular prognathism. Severe hemorrhage ensued while completing the osteotomy on the left side. The vessel involved was not specifically mentioned, but the bleeding came from an area anterior to the posterior border of the mandible, approximately midway up the ascending ramus. The bleeding was stopped by pressure and the patient’s initial postoperative course was uneventful. Nine weeks after surgery, he developed a preauricular pulsatile swelling. Angiography confirmed the presence of a pseudoaneurysm, probably of the maxillary artery, which was successfully embolized with a Gianturco (Cook, Bloomington, IN) coil. Hemmig et al2 reported a ruptured pseudoaneurysm of the sphenopalatine artery postoperatively in a 29-year-old woman who underwent a Le Fort I osteotomy with impaction to correct vertical maxillary excess. In the postoperative period, she developed recurrent epistaxis that was not controlled by anterior and posterior nasal packing. Angiography showed a pseudoaneurysm of the sphenopalatine artery that was successfully embolized by a Gianturco coil. Solomons and Blumga& reported a case of a 20-year-old man with severe, lifethreatening, left-sided epistaxis 1 month after a Le Fort I osteotomy to correct a malocclusion. Angiography revealed a false aneurysm of the left maxillary artery posterior to the left maxillary antrum. The aneurysm was successfully embolized by a large Gelfoam (Upjohn, Don Mills, Ontario, Canada) pledget followed by a 3-mm Gianturco coil. Lustbader et al8 reported a case of severe epistaxis, 12 days postoperatively, in a 17-year-old boy who had undergone a Le Fort I osteotomy. Angiography demonstrated a false aneurysm of the maxillary artery that was successfully embolized with three guide wires. Four cases of A-V listula involving the internal
573
LANIGAN ET AL
carotid artery system following orthognathic surgery have been reported.3-5*7 Haba13 reported a case of carotid-cavernous sinus fistula after a modified Le Fort II osteotomy in a 28-year-old woman with Charcot-Marie-Tooth syndrome. Lanigan and Tubman reported a carotid-cavernous sinus fistula in a 23-year-old man with vertical maxillary excess treated by a Le Fort I osteotomy intrusion. A small false aneurysm of the inferior aspect of the internal carotid artery was also noted. Hes and de Man’ reported a carotid-cavernous sinus tistula postoperatively in a 19-year-old man with a midfacial retrusion secondary to a cleft lip and palate deformity who underwent a Le Fort I advancement osteotomy. In all three cases the carotid-cavernous tistulas were successfully obliterated by balloon embolization. Newhouse et al’ reported a traumatic A-V fistula involving the internal carotid artery and internal jugular vein at the base of the skull following a Le Fort I osteotomy with intrusion to correct vertical maxillary excess. The internal carotid artery was subsequently ligated in the neck and the jugular foramen was obliterated with a muscle graft. Report of Cases In 1986, a questionnaire pertaining to major vascular complications following orthognathic surgery was sent to every oral and maxillofacial surgeon in North America. Five thousand questionnaires were sent and almost 800 replies were received. Five cases of false aneurysm and A-V tistula not previously reported in the literature were received in response to our questionnaire. Adequate documentation exists for four of the cases, which are discussed in the following case histories. The fifth case, involving a false aneurysm of the maxillary artery following a vertical subcondylar osteotomy for mandibular prognathism, was not included because full documentation was unobtainable. Case I A 13-year-old girl with vertical maxillary excess and mandibular retrognathia was treated by a Le Fort I osteotomy with intrusion and a mandibular advancement. Her initial postoperative course was uneventful. Six weeks postoperatively, however, she experienced massive epistaxis resulting in hemorrhagic shock. She was resuscitated in a local hospital with 4 L of intravenous fluid, but no blood products, and was then transferred to the hospital where her initial surgery had been performed. The patient was actively bleeding from the right nostril and back into the pharynx when seen in the emergency room. She was immediately removed from maxillomandibular fixation to ensure a more adequate airway. She was transferred to the intensive care unit (ICU) and an anterior nasal pack was placed. As the anterior nasal pack did not stop the bleeding, a posterior nasal pack using a Foley
catheter and gauze was also placed. This arrested the hemorrhage. Her hemoglobin on admission was 8.3 g/dL, her hematocrit was 25.5%, and her prothrombin time (ET) was 16 seconds as compared to a control level of 11.5 seconds. She was given 4 U of fresh-frozen plasma and 6 U of packed red blood cells. She also was given vitamin K 10 mg once per day for 4 days. Her hemoglobin on the second hospital day was 12.1 g/dL, with a hematocrit of 36%. On the third day following admission, she underwent a right carotid artery arteriogram that revealed a false aneurysm, approximately 1 cm in diameter, in the right maxillary sinus. The pseudoaneurysm appeared to be from a distal branch of the maxillary artery, probably the sphenopalatine artery. On the fifth day, she was taken back to the operating room. Her hemoglobin preoperatively was 10.8 g/dL, with a hematocrit of 32.6%. The right external carotid artery was ligated in the neck. A Le Fort I maxillary osteotomy with downfracture was then performed. The false aneurysm was easily visualized, found to be intact, and was resected. The right maxillary artery was then ligated by a transantral approach from the downfracture position. A small amount of Surgicel (Johnson & Johnson, New Brunswick, NJ) was placed in the posterior maxillary region. Intraosseous and suspension wires were used to fix the maxilla back into position, but no maxillomandibular fixation was used. The estimated blood loss during the Le Fort I procedure was 200 mL. The patient received 1 U of packed red blood cells and 2 U of fresh frozen plasma. The postoperative hemoglobin was 9.6 g/dL. with a hematocrit of 29%. Her nasal packs were removed 2 days postoperatively and, after no active bleeding was noted, she was discharged on the fourth postoperative day. The rest of her postoperative course was uneventful.
Case 2 A 20-year-old woman with vertical maxillary excess and relative mandibular retrognathia was treated with a Le Fort I osteotomy with intrusion and slight retrusion, and an advancement genioplasty. No intraoperative complications were noted and the estimated blood loss at surgery was less than 500 mL. Minor hemorrhage intraoperatively from the left descending palatine artery was controlled with a hemoclip. Her initial postoperative course was uneventful and she was discharged on the second postoperative day. Three weeks following surgery, she had her first episode of bleeding from the right nose. As it had ceased spontaneously by the time she was seen in the emergency room, she was discharged with no treatment. She continued, however, to have intermittent epistaxis from the right nose. Four and one-half weeks postoperatively she had her second significant episode of bleeding, although it again ceased spontaneously. She was subsequently referred to an ENT surgeon for endoscopic examination of the right nose. No active bleeding was noted at that time, although some granulation tissue at the inferior posterior aspect of the inferior turbinate was cauterized. No nasal packing was used, but she was placed on mild sedation and bedrest. No further bleeding episodes were noted until 1 week following the cauterization, 7 weeks postoperatively, when the patient had to be readmitted to hospital for right-sided epistaxis. The patient was taken to the operating room where under intravenous sedation an Epistat
574
ORTHOGNATHIC
catheter with a IO-mL balloon was placed in the right nose and inflated to act as a posterior nasal pack. An anterior nasal pack was placed using Y4-in gauze with Terramycin (Pfizer, New York, NY) ointment. As no further bleeding occurred, the patient was discharged the next morning. Her hemotocrit on admission was 28%, but no blood transfusion was given at that time. Five days following her discharge she had to be readmitted. At 6:00 AM she had experienced fullness in her right cheek and right-sided posterior epistaxis into the pharynx. She was not actively bleeding when seen initially in the emergency department, so her anterior nasal pack was removed, followed by the posterior nasal pack. No hemorrhage was encountered from the nose: however, a small amount of oozing was noted from the Le Fort I incision site in the right maxillary vestibule. Blood also could be expressed from the incision site near the first molar area with compression of the right cheek. On admission the patient’s hemoglobin was 7.8 g/dL, the hematocrit was 26%, platelets were 346,000/mm3, prothrombin time (ET) was 12 seconds, partial thromboplastin time (ETT) was 30 seconds, fibrinogen was 335 mg%, fibrin split products were not detectable, and the ristocetin factor VIII cofactor was 1.13. All coagulation factors appeared normal except for the Ivy bleeding time, which was at the high range of normal at 9.5 minutes (range, 2.5 to 9.5 minutes). The patient had not been on any nonsteroidal anti-inflammatory drugs, and her family history and past history were negative for bleeding problems. The hematologist who saw her in consultation did not feel her bleeding was secondary to a coagulopathy. He suggested she be given 2 U of packed red blood cells and advised placing her on prophylactic Amicar (aminocaproic acid; Lederle, Markham, Ontario, Canada) 4 g every 4 hours for 7 days. No further bleeding episodes occurred until 11 days later, which was 11 weeks following her original orthognathic surgery. The patient on awakening in the morning had noticed some swelling in the right cheek and subsequently had a small amount of bleeding from the mouth and nose, which stopped spontaneously. An hour later, however, more vigorous bleeding commenced, although this was stopped temporarily with two squirts of Afrin (oxymetazoline hydrochloride; Schering, Kenilworth, NJ) nasal spray into her right nostril. The patient was readmitted to hospital as she continued to experience some epistaxis. She underwent a right carotid arteriogram that revealed a 9 x 13-mm vascular malformation that was apparently a pseudoaneurysm of the sphenopalatine branch of the maxillary artery. Selective embolization of this malformation could not be carried out, however, because catheterization of the distal aspect of the maxillary artery could not be accomplished because of spasm of the maxillary artery. The patient was actively bleeding at the time of the arteriogram. Because of poor control of the bleeding it was decided to take the patient back to the operating room. In conjunction with an ENT surgeon, a transantral approach to ligate the right maxillary artery was attempted. This was unsuccessful because of altered anatomy and marked hemorrhage which resulted in poor visualization. The right external carotid artery was therefore ligated in the neck above the lingual artery. This signit’icantly decreased the bleeding, which was then stopped by additional packing in the posteriomedial aspect of the maxilla with strips of Surgicel. The patient received an additional 2 U of whole blood to increase her hematocrit to 34% at
SURGERY-FALSE
ANEURYSMS
AND A-V FISTULAS
the time of discharge. She was placed on prophylactic Amicar 4 g every 4 hours for 2 weeks at the suggestion of the hematologist. No other active treatment was carried out at that time other than a continuation of her limitation of physical activity. The neuroradiologist was consulted about the feasibility of selective embolization of the contralateral maxillary artery due to concerns about revascularization of the right maxillary artery occurring from the left side. The neuroradiologist advised against this because he felt there was a possibility of dislodging the clot from the right maxillary artery during the arteriograms. The patient fortunately had no further bleeding episodes. Case 3 A 29-year-old man with maxillary retrusion secondary to a previous Le Fort II fracture in a motor vehicle accident 2 years previously was treated with a Le Fort I maxillary osteotomy with advancement. Slight nonspecific bleeding was noted from the left posterior maxillary region at the time of surgery. The estimated blood loss at surgery was 1,000 mL and the patient received 1 U of autologous packed red blood cells intraoperatively. His initial course was uneventful until 6 days postoperatively when, at approximately 5:OOPM, he developed intractable bleeding from his nose and mouth, especially the left nostril. The bleeding had been brought on when he blew his nose following exercising by lifting weights. The patient was taken to the emergency department at a local hospital where it took 4 hours to bring the bleeding under control. The bleeding slowed significantly after an ENT surgeon placed left anterior and posterior nasal gauze packs and a balloon catheter as a right posterior nasal pack. The nose bleed was persistent through the evening, but by early the next morning the epistaxis had significantly decreased and the patient was considered stable; no bleeding was noted going back down into the pharynx. On the second day after the bleeding had commenced the patient was transfused with 4 U of packed red blood cells because of his low hemoglobin. The nasal packs were left in place for a total of 5 days, and the patient was hospitalized for 1 week in total. Two days following his discharge he was admitted to a larger medical center for arteriograms. The patient was not experiencing any active bleeding at that time. A neuroradiologist performed arteriography of the left and right external carotid artery systems. A small false aneurysm of the left maxillary artery was noted. The neuroradiologist embolized both the left and right maxillary and facial arteries in an attempt to prevent any recurrence of the epistaxis. The patient was discharged from hospital the following day. The rest of the patient’s postoperative course was uneventful, with no long-term sequelae.
Case 4 A 27-year-old woman with a midfacial retrusion secondary to a left unilateral cleft lip and palate deformity underwent a Le Fort I osteotomy with advancement and a bone graft to the alveolar cleft site. The surgeon encountered difficulty in downfracturing the maxilla due to very thick bone, especially along the posterior maxillary walls. Excessive generalized bleeding was noted intraoperatively, but no specific vessel could be identified as the source of the hemorrhage. The patient received 3 U of packed red blood cells intraoperatively. Postoperatively,
LANIGAN
575
ET AL
in the recovery room, the patient was noted to have chemosis of the left eye, mild left ptosis, diplopia, and an inability to move the left eye laterally from the midline due to a left VI nerve palsy. A computed tomography (CT) scan was reported as normal, but a left carotid arteriogram demonstrated an extensive left carotidcavernous fistula. The patient was referred to another institution where successful balloon embolization of the carotid-cavernous fistula was carried out. The patient’s double vision gradually resolved as the VI nerve palsy improved. By 6 months postoperatively her vision was normal, the ptosis had resolved, and the “pulsating pressure” that she once had in the temporal region had disappeared. The patient, however, continued to complain of mild pain in the maxillary region bilaterally.
Discussion From a review of the literature, and from the cases presented in this report, it is possible to draw several conclusions regarding false aneurysms and A-V listulas following orthognathic surgery. False aneurysms following mandibular orthognathic surgery, especially vertical or oblique mandibular ra-
mus osteotomies, are most likely to involve the internal maxillary artery,’ which is usually lacerated when the osteotomy cut is made near the sigmoid notch.26 Care must be taken to avoid saws that cut so effectively that they not only section the bone well, but also cut through into the soft tissues along the medial side of the ascending ramus. Marked bleeding is usually encountered at the time of surgery, which may be satisfactorily controlled intraoperatively by pressure packing. Signs of a false aneurysm, such as an enlarging pulsatile mass or recurrent bleeding, may eventually become obvious. The vessel most commonly involved with false aneurysms following maxillary orthognathic surgery is the internal maxillary artery, in particular the sphenopalatine branch. The internal maxillary artery and its branches are most vulnerable to damage in their location in the pterygopalatine fossa when the maxillary tuberosity is separated from the pterygoid plates with an osteotome27~28 or during the downfracture procedure.29 The classical signs of a false aneurysm, such as a bruit or palsatile expanding mass, may not be evident from vessels damaged during a Le Fort I osteotomy due to their deep location. The first indication of the problem may be persistent or recurrent epistaxis that does not respond to measures such as anterior and posterior nasal packing. One factor that may be unusual in their presentation is the time at which the initial bleeding first occurs. In the series of cases of hemorrhage following Le Fort I osteotomies previously reported, 29.30the vast majority of the initial episodes occurred within the first 14 days postoperatively. The initial bleeding associated with a false aneurysm can present later than this, how-
ever. The patient in case 1 experienced her first episode of significant epistaxis 6 weeks postoperatively, whereas the patient in case 2 had her initial episode of bleeding 3 weeks after surgery. The patient reported by Solomons and Blumga& first presented with epistaxis 1 month postoperatively. Ideally, any patient who presents with the first episode of epistaxis after the second postoperative week, or who experiences recurrent significant hemorrhage following orthognathic surgery, should be investigated by angiography to rule out a false aneurysm as a source of the hemorrhage. If the patient is actively bleeding at the time of the investigation, angiography may be able to locate the actual site of the hemorrhage. Angiography also helps in delineating the development of collateral blood flow to an area, and whether or not the contralateral arterial tree is contributing to the problem.3’ If a false aneurysm is diagnosed during angiography, the treatment of choice is transcatheter embolization.“,6 Surgery can also be used to resect the false aneurysm (see case l), but this treatment should only be used if it can be done safely without placing adjacent structures at risk, or risking rupture of the false aneurysm.” Schwartz reported a case” of a false aneurysm of the internal maxillary artery following Le Fort II and mandibular fractures. Embolization failed to control the aneurysm and its size and location made a direct surgical approach to resect it unsafe. Surgery was therefore performed to clip the third portion of the internal maxillary artery via a transantral approach, as well as to ligate the distal branches of the external carotid artery that might provide collateral flow to the lesion, including the common facial-lingual trunk, and the ascending pharyngeal, occipital, and superficial temporal arteries. One of the advantages of embolization over surgical intervention is that the more distal vessels that supply the bleeding source may be obliterated while sparing the more proximal vessels.3’ This is particularly important following orthognathic surgery where one wishes to avoid a further compromise of an already diminished vascular supply, which could contribute to the development of aseptic necrosis of the maxilla.33 Selective embolization as distally as possible will also minimize the risk of persistent hemorrhage from collateral flow. With false aneurysms, embolization with agents such as small pieces of Gelfoam may be unsuccessful and devices such as Gianturco coils may have to be used.2,6*8 Arteriovenous fistulas following orthognathic surgery are more apt to involve large vessels, especially the internal carotid artery.3-5’7 The basis for injuries to the internal carotid artery following orthognathic surgery has been discussed by Lani-
576
ORTHOGNATHIC
gan.25 Direct trauma to the artery can occur during the pterygomaxillary dysjunction or the maxillary downfracture when a basal skull fracture ensues that involves areas such as the foramen lacerum or the carotid canal. Indirect trauma can follow sudden forceful stretching of the internal carotid artery in the neck over the upper cervical spine by cervical hyperextension with contralateral rotation of the head. A severe disruption of the layers of the wall of the internal carotid artery could lead to the formation of an A-V listula. A carotid-cavernous fistula may be clinically obvious early in the postoperative course,7 or the development of signs and symptoms may be delayed for weeks or months.3*4 The diagnosis can be confirmed by carotid angiography. In all the cases of carotid-cavernous sinus fistula following orthognathic surgery reported to date3,4*7 (case 4), the tistulas have been successfully obliterated by detachable balloon catheters, with no recurrence. Matching the size of the embolic material to the shunt is critical for success with this procedure. The advantage of the balloon catheter technique over techniques used in the past is that it can obliterate the tistula more safely, while preserving blood flow in the internal carotid artery.4*7 Because embolization may not totally seal the shunt in an A-V tistula, there is always the risk that the defect may gradually reopen. 24 Embolization may be repeated, if necessary, however. The occurrence of direct trauma to the carotid artery secondary to basal skull fractures would appear to be related to difficult downfractures of the maxilla4 (case 4). If a difftcult downfracture is encountered following completion of routine Le Fort I osteotomies, then the posterior walls of the maxilla should be sectioned with an osteotome or, alternatively, sectioning to achieve the pterygomaxillary dysjunction can be attempted through the maxillary tuberosity rather than between the tuberosity and pterygoid plates34 in an attempt to avoid, as much as possible, the thick posterior maxillary walls. If a relatively atraumatic downfracture still cannot be achieved following these maneuvers, consideration should be given to stopping the procedure and obtaining a CT scan to look at the posterior maxilla/pterygoid plate regions. If the CT scan shows excessive thickness of bone in this region, or abnormalities in the base of the skull that could increase the relative risk of an injury to the internal carotid artery system or of hemorrhage from damage to the maxillary artery or its branches, then the surgeon may wish to discuss these possibilities further with the patient prior to proceeding with additional surgery. In certain patients with craniofacial malformations it may be prudent to do the
SURGERY-FALSE
ANEURYSMS
AND A-V FISTULAS
CT scan even before proceeding with the initial surgery. 35 Surgeons should also be aware of the possibility that congenital A-V malformations that could lead to marked intraoperative and/or postoperative hemorrhage might be encountered while performing orthognathic surgery. Lanigan et al*‘j reported a case of major intraoperative bleeding from a congenital A-V malformation that was lacerated along the medial surface of the mandible when an intraoral vertical mandibular ramus osteotomy was performed for unilateral condylar hyperplasia. Acknowledgment The authors would like to thank the surgeons who responded to our inquires, often on more than one occasion, for details regarding their specific cases. We would also like to thank the patient in case 3 who also responded to our inquiry with additional information regarding his complication.
References I. Clark R, Lew D, Giyanani VL, et al: False aneurysm com2.
3. 4.
5.
6.
7.
8.
9. 10.
11.
12. 13. 14. 15.
16.
plicating orthognathic surgery. J Oral Maxillofac Surg 4557, 1987 Hemmig S, Johnson RS, Ferraro N: Management of a ruptured pseudoaneurysm of the sphenopalatine artery following a Le Fort I osteotomy. J Oral Maxillofac Surg 45533, 1987 Habal M: A carotid cavernous sinus fistula after maxillary osteotomy. Plast Reconstr Surg 77:981, 1986 Lanigan D, Tubman D: Carotid-cavernous sinus fistula following orthognathic surgery. J Oral Maxillofac Surg 45:%9, 1987 Newhouse R, Schow S, Kraut R, et al: Life-threatening hemorrhage from a Le Fort I osteotomy. J Oral Maxillofat Surg 40: 117, 1982 Solomons NB, Blumgart R: Severe late-onset epistaxis following Le Fort I osteotomy: Angiographic localization and embolization. J Laryngol Otol 102:260, 1988 Hes J, de Man K: Carotid-cavernous sinus Iistula following maxillofacial trauma and orthognathic surgery. Int J Oral Maxillofac Surg 17:295, 1988 Lustbader D, Schwartz M, Zito J, Stem M: The use of percutaneous transcatheter embolization to control postoperative bleeding following Le Fort I osteotomy: Report of three cases. J Oral Maxillofac Surg (accepted) D’Orta J, Shatney CH: Post-traumatic pseudoaneurysm of the internal maxillary artery. J Trauma 22: 161, 1982 Field PL, Tress BM, O’Brien MD, et al: Embolization of traumatic aneurysm of the maxillary artery. Aust NZ Surg 49:244, 1979 Callem WS: Traumatic (false) aneurysms of the terminal portion of the external carotid artery. Am J Surg 106:522, 1963 Bresner M, Brekke J, Dubit J, et al: False aneurysms of the facial region. J Oral Surg 30:307, 1972 Wineland PL. Topazian RG, Marble HB: False aneurysm of the facial artery. J Oral Surg 34642, 1976 Tatelman M: Arteriography in oral and maxilofacial surgery. J Oral Surg 34:795, 1976 Marks MW, Argenta LC, Dingman RO: Traumatic arteriovenous malformation of the external carotid arterial system. Head Neck Surg 6:1054, 1984 Guzman Freire JE, Cabanas Valls R, Munoz Noya JA, et al: Arteriovenous fistulas of the anterior facial vessels: Review of the literature and report of case. J Oral Surg 32: 150, 1974
LANIGAN
577
ET AL
17. Cooperband B. Friedel W, Bhatt GM, et al: False aneurysm of the facial artery. J Oral Maxillofac Surg 47: 1327, 1989 18. Taylor DV: Traumatic aneurysm and facial palsy as a complication of a mandibular fracture. Br J Oral Surg 4:202, I%7 19. Schwartz H. Kendrick R, Pogorel B: False aneurysm of the maxillary artery. An unusual complication of closed facial trauma. Arch Otolaryngol 109:616, 1983 20. Van den Akker HP, Van der Lijn FA: A false aneurysm of the facial artery as a complication of circumferential wiring. Oral Surg 37514, 1974 21. Miller FJ, Entzminger LB, Coleman LL, et al: Successful catheter embolization of a false aneurysm of the internal maxillary artery. Arch Otolaryngol 101:517, 1975 22. Peoples JR, Herbosa EG, Dion J: Management of internal maxillary artery hemorrhage from temporomandibular joint surgery via selective embolization. J Oral Maxillofac Surg 46: 1005, 1988 23. Bradley P: Conservative treatment for temporomandibular joint pain dysfunction. Br J Oral Maxillofac Surg 25: 125. 1987 24. Brown JS, Ward-Booth RP: Arterio-venous malformation following a radical neck dissection. Int J Oral Maxillofac Surg 18:239, 1989 25. Lanigan DT: Injuries to the internal carotid artery following orthognathic surgery. Int J Adult Ortho Orthognath Surg 4:215. 1988 26. Lanigan DT. Hey JH. West RA: Hemorrhage following man-
dibular osteotomies. A report of twenty-one cases. J Oral Maxillofac Surg (accepted) 27. Turvey T, Fonseca R: The anatomy of the maxillary artery in the pterygopalatine fossa: Its relationship to maxillary surgery. J Oral Surg 38:92, 1980 28. Robinson P, Hendy C: Pterygoid plate fractures caused by the Le Fort I osteotomy. Br J Oral Maxillofac Surg 24:198, 1986 29. Lanigan DT, Hey JH, West RA: Major vascular complications of orthognathic surgery: Hemorrhage associated with Le Fort I osteotomies. J Oral Maxillofac Surg 48:561.
1990 30. Lanigan DT, West RA: Management of postoperative hemorrhage following the Le Fort I maxillary osteotomy. J Oral Maxillofac Surg 42:367, 1984 3 1. Schilstra S, Marsman J: Embolization for traumatic epistaxis. J Craniomaxillofac Sure. 15:28. 1987 32. Kingsley D, O’Conner A: Embolization in otolaryngology. J Laryngol Otol 96:439, 1982 33. Lanigan DT. Hey JH. West RA: Aseptic necrosis of the maxilla. Report of 36 cases. J Oral Maxillofac Surg 48:142, 1990 34. Timble LD. Tideman H, Stoelinga P: A modification of the pterygoid plate separation in low-level maxillary osteotomies. J Oral Maxillofac Surg 41:544, 1983 35. Reaume C, MacNichol B: Complications encountered during Le Fort I osteotomy in a patient with mandibulofacial dysostosis. J Oral Maxillofac Surg 46:1003. 1988
Surg
1991
Major Vascular Complications Orthogna thic Surgery:
of
False Aneurysms and Arteriovenous Fistulas Following Orthogna thic Surgery DENNIS T. LANIGAN, DMD, MD,* JULIANA H. HEY, DDS, MD,t AND ROGER A. WEST, DMDS False aneurysms and arteriovenous fistulas are rare complications of orthognathic surgery. The vessel most commonly involved with false aneurysms following mandibular surgery is the internal maxillary artery, and this vessel, especially the sphenopalatine branch, may also be involved following maxillary surgery. An unusual factor in the presentation of false aneurysms following Le Fort I osteotomies is an initial episode of epistaxis occurring greater than 2 weeks postoperatively. Arteriovenous fistulas following orthognathic surgery are more apt to involve large vessels, especially the internal carotid artery. Embolization procedures are the treatment of choice for false aneurysms and arteriovenous fistulas in the maxillofacial region following orthognathic surgery.
aneurysm usually results from a tangential, incomplete tear of the arterial vessel wall so that the continuity of blood flow through the lacerated artery is generally maintained.13 Hemorrhage occurs into the adjacent soft tissues surrounding the artery until hematoma formation compresses the area and tamponades the bleeding. The hemorrhage continues until pressure in the periarterial zone equals the mean arterial pressure.’ The hematoma subsequently organizes at the periphery, with the perivascular connective tissue creating a sac around which an endothelial lining forms to produce a pseudointima that remains in continuity with the arterial lumen.**14 With liquefaction of the hematoma, a communication develops between the artery and the aneurysmal sac resulting in a pulsating mass. The arterial pressure leads to gradual expansion of the false aneurysm, which can either rupture or continue to progressively enlarge.‘,‘3*‘9 An incomplete laceration of an artery in conjunction with a concomitant injury to its accompanying vein can lead to an abnormal communication between them, resulting in an A-V tistula. The incomplete disruption of the arterial wall results in hemorrhage and hematoma formation that eventually tamponades the bleeding. The mechanism for the development of a traumatic A-V fistula has been postulated to be the organization of a hematoma
False aneurysms (pseudoaneursysms) and arteriovenous (A-V) fistulas have only rarely been reported following orthognathic surgery,le8 facial trauma,““’ or other surgical procedures in the head and neckmaxillofacial region.‘2-24 The small size of most blood vessels in the facial region makes their partial transection unlikely, and this is the principal reason for the rarity of false aneurysms or A-V fistulas in this locationg3” Penetrating or blunt trauma can lead to arterial damage. A traumatic intimal laceration can act as the site for occlusion of the vessel or for the origin of a false aneurysm. A more severe disruption of the arterial wall could lead to hemorrhage or the formation of an A-V fistula.4*25 A false
* Professor and Head, Division of Oral and Maxillofacial Surgery, University of Saskatchewan. t Formerly Clinical Fellow, Division of Oral and Maxillofacial Surgery, University of Saskatchewan; currently, Clinical Instructor, College of Dentistry, University of Saskatchewan. $ Director, Northwest Center for Corrective Jaw Surgery, Seattle. Address correspondence and reprint requests to Dr Lanigan: Division of Oral and Maxillofacial Surgery, College of Dentistry, University of Saskatchewan, Saskatoon, Saskatchewan, S7N OWOCanada. 0 1991 geons 0278-2391
American
Association
of Oral
and Maxillofacial
Sur-
I91 /4906-0004$3.00/O
571
572
ORTHOGNATHIC
arising from damage to the vaso vasorum of the artery. 24 Endothelial bud proliferation into the organizing hematoma can lead to multiple endothelial lined channels forming between the damaged arterial and venous systems rather than just a single communication.” A listula may be established immediately, or it may not become apparent for days or weeks. A shunting of blood from the arterial to the venous system results in a bypassing of the capillary system resistance and peripheral blood flow is decreased in the involved artery. The decreased vascular resistance at the site of the flstula leads to blood flow tending to preferentially enter the A-V fistula and to the progressive development of an extensive collateral circulation around the fistula. The increased blood flow to the area produces a dilation of the feeding arteries, while increased pressure in the draining veins results in them becoming dilated and thickened.15 An A-V fistula can produce a continuous vibratory thrill and a bruit, which is intensified during systole. There is wide transmission of a “machinery-like” murmur.t2*t3 With false aneurysms, depending on the amount of thrombus within the aneurysmal sac, the mass could be pulsatile and a bruit may be present. l9 The bruit is generally heard in systole and remains localized. I2 With A-V Iistulas there can be an absence or a more localized degree of swelling than is seen with the continuously expanding sac of a false aneu12,13 The abundant collateral circulation that rysm. develops with an A-V Iistula is in contrast to the poor collateral circulation found with the false aneurysm. I2 The dilation and pulsation of local veins that can be seen in an A-V listula are not seen with false aneurysms. Untreated false aneurysms may progressively enlarge until they rupture, leading to significant hemorrhage. l3 Enlargement without rupture can lead to facial asymmetry if the lesion is located near the surface of the skin. Pressure on nerves from the enlargement of a pseudoaneurysm also can result in pain or paresis.15 On the other hand, a localized A-V fistula can be relatively indolent, with no progressive enlargement or tendency to rupture. l2 On radiographic examination, false aneurysms are characterized by residual contrast in the lesion long after the arterial phase of angiography is completed.’ False aneurysms can be caused by blunt’2,13*17or, more commonly, by penetrating trauma. Gunshot wounds or knife injuries account for the majority of cases9*13 although vessels can also be lacerated by facial fractures’4*‘8’19’21 or the placement of a circumandibular wire. 2o The superficial temporal and facial arteries are the vessels in the head and neck region most commonly involved in traumatic false aneurysms. l1 The superficial temporal artery is es-
SURGERY-FALSE
ANEURYSMS
AND A-V FISTULAS
pecially vulnerable to injury due to its long and superficial course.9 The relatively small size, and deep, well-protected, location of the maxillary artery undoubtedly contribute to the rare occurrence of posttraumatic pseudoaneurysms in this vessel. In the posttraumatic cases that have been reported, two cases were related to penetrating trauma,‘.” one to compound fractures of the mandibular ramus and condylar neck,” and one to a Le Fort II and mandibular fracture.” Miller et a121reported a case of a large false aneurysm from the sphenopalatine branch of the maxillary artery in a young man who developed severe epistaxis following a Le Fort III fracture of the maxilla. Four cases of false aneurysm following orthognathic surgery have been reported to date. Clark et al’ reported the case of a 15year-old boy who underwent bilateral mandibular vertical subcondylar osteotomies for the correction of mandibular prognathism. Severe hemorrhage ensued while completing the osteotomy on the left side. The vessel involved was not specifically mentioned, but the bleeding came from an area anterior to the posterior border of the mandible, approximately midway up the ascending ramus. The bleeding was stopped by pressure and the patient’s initial postoperative course was uneventful. Nine weeks after surgery, he developed a preauricular pulsatile swelling. Angiography confirmed the presence of a pseudoaneurysm, probably of the maxillary artery, which was successfully embolized with a Gianturco (Cook, Bloomington, IN) coil. Hemmig et al2 reported a ruptured pseudoaneurysm of the sphenopalatine artery postoperatively in a 29-year-old woman who underwent a Le Fort I osteotomy with impaction to correct vertical maxillary excess. In the postoperative period, she developed recurrent epistaxis that was not controlled by anterior and posterior nasal packing. Angiography showed a pseudoaneurysm of the sphenopalatine artery that was successfully embolized by a Gianturco coil. Solomons and Blumga& reported a case of a 20-year-old man with severe, lifethreatening, left-sided epistaxis 1 month after a Le Fort I osteotomy to correct a malocclusion. Angiography revealed a false aneurysm of the left maxillary artery posterior to the left maxillary antrum. The aneurysm was successfully embolized by a large Gelfoam (Upjohn, Don Mills, Ontario, Canada) pledget followed by a 3-mm Gianturco coil. Lustbader et al8 reported a case of severe epistaxis, 12 days postoperatively, in a 17-year-old boy who had undergone a Le Fort I osteotomy. Angiography demonstrated a false aneurysm of the maxillary artery that was successfully embolized with three guide wires. Four cases of A-V listula involving the internal
573
LANIGAN ET AL
carotid artery system following orthognathic surgery have been reported.3-5*7 Haba13 reported a case of carotid-cavernous sinus fistula after a modified Le Fort II osteotomy in a 28-year-old woman with Charcot-Marie-Tooth syndrome. Lanigan and Tubman reported a carotid-cavernous sinus fistula in a 23-year-old man with vertical maxillary excess treated by a Le Fort I osteotomy intrusion. A small false aneurysm of the inferior aspect of the internal carotid artery was also noted. Hes and de Man’ reported a carotid-cavernous sinus tistula postoperatively in a 19-year-old man with a midfacial retrusion secondary to a cleft lip and palate deformity who underwent a Le Fort I advancement osteotomy. In all three cases the carotid-cavernous tistulas were successfully obliterated by balloon embolization. Newhouse et al’ reported a traumatic A-V fistula involving the internal carotid artery and internal jugular vein at the base of the skull following a Le Fort I osteotomy with intrusion to correct vertical maxillary excess. The internal carotid artery was subsequently ligated in the neck and the jugular foramen was obliterated with a muscle graft. Report of Cases In 1986, a questionnaire pertaining to major vascular complications following orthognathic surgery was sent to every oral and maxillofacial surgeon in North America. Five thousand questionnaires were sent and almost 800 replies were received. Five cases of false aneurysm and A-V tistula not previously reported in the literature were received in response to our questionnaire. Adequate documentation exists for four of the cases, which are discussed in the following case histories. The fifth case, involving a false aneurysm of the maxillary artery following a vertical subcondylar osteotomy for mandibular prognathism, was not included because full documentation was unobtainable. Case I A 13-year-old girl with vertical maxillary excess and mandibular retrognathia was treated by a Le Fort I osteotomy with intrusion and a mandibular advancement. Her initial postoperative course was uneventful. Six weeks postoperatively, however, she experienced massive epistaxis resulting in hemorrhagic shock. She was resuscitated in a local hospital with 4 L of intravenous fluid, but no blood products, and was then transferred to the hospital where her initial surgery had been performed. The patient was actively bleeding from the right nostril and back into the pharynx when seen in the emergency room. She was immediately removed from maxillomandibular fixation to ensure a more adequate airway. She was transferred to the intensive care unit (ICU) and an anterior nasal pack was placed. As the anterior nasal pack did not stop the bleeding, a posterior nasal pack using a Foley
catheter and gauze was also placed. This arrested the hemorrhage. Her hemoglobin on admission was 8.3 g/dL, her hematocrit was 25.5%, and her prothrombin time (ET) was 16 seconds as compared to a control level of 11.5 seconds. She was given 4 U of fresh-frozen plasma and 6 U of packed red blood cells. She also was given vitamin K 10 mg once per day for 4 days. Her hemoglobin on the second hospital day was 12.1 g/dL, with a hematocrit of 36%. On the third day following admission, she underwent a right carotid artery arteriogram that revealed a false aneurysm, approximately 1 cm in diameter, in the right maxillary sinus. The pseudoaneurysm appeared to be from a distal branch of the maxillary artery, probably the sphenopalatine artery. On the fifth day, she was taken back to the operating room. Her hemoglobin preoperatively was 10.8 g/dL, with a hematocrit of 32.6%. The right external carotid artery was ligated in the neck. A Le Fort I maxillary osteotomy with downfracture was then performed. The false aneurysm was easily visualized, found to be intact, and was resected. The right maxillary artery was then ligated by a transantral approach from the downfracture position. A small amount of Surgicel (Johnson & Johnson, New Brunswick, NJ) was placed in the posterior maxillary region. Intraosseous and suspension wires were used to fix the maxilla back into position, but no maxillomandibular fixation was used. The estimated blood loss during the Le Fort I procedure was 200 mL. The patient received 1 U of packed red blood cells and 2 U of fresh frozen plasma. The postoperative hemoglobin was 9.6 g/dL. with a hematocrit of 29%. Her nasal packs were removed 2 days postoperatively and, after no active bleeding was noted, she was discharged on the fourth postoperative day. The rest of her postoperative course was uneventful.
Case 2 A 20-year-old woman with vertical maxillary excess and relative mandibular retrognathia was treated with a Le Fort I osteotomy with intrusion and slight retrusion, and an advancement genioplasty. No intraoperative complications were noted and the estimated blood loss at surgery was less than 500 mL. Minor hemorrhage intraoperatively from the left descending palatine artery was controlled with a hemoclip. Her initial postoperative course was uneventful and she was discharged on the second postoperative day. Three weeks following surgery, she had her first episode of bleeding from the right nose. As it had ceased spontaneously by the time she was seen in the emergency room, she was discharged with no treatment. She continued, however, to have intermittent epistaxis from the right nose. Four and one-half weeks postoperatively she had her second significant episode of bleeding, although it again ceased spontaneously. She was subsequently referred to an ENT surgeon for endoscopic examination of the right nose. No active bleeding was noted at that time, although some granulation tissue at the inferior posterior aspect of the inferior turbinate was cauterized. No nasal packing was used, but she was placed on mild sedation and bedrest. No further bleeding episodes were noted until 1 week following the cauterization, 7 weeks postoperatively, when the patient had to be readmitted to hospital for right-sided epistaxis. The patient was taken to the operating room where under intravenous sedation an Epistat
574
ORTHOGNATHIC
catheter with a IO-mL balloon was placed in the right nose and inflated to act as a posterior nasal pack. An anterior nasal pack was placed using Y4-in gauze with Terramycin (Pfizer, New York, NY) ointment. As no further bleeding occurred, the patient was discharged the next morning. Her hemotocrit on admission was 28%, but no blood transfusion was given at that time. Five days following her discharge she had to be readmitted. At 6:00 AM she had experienced fullness in her right cheek and right-sided posterior epistaxis into the pharynx. She was not actively bleeding when seen initially in the emergency department, so her anterior nasal pack was removed, followed by the posterior nasal pack. No hemorrhage was encountered from the nose: however, a small amount of oozing was noted from the Le Fort I incision site in the right maxillary vestibule. Blood also could be expressed from the incision site near the first molar area with compression of the right cheek. On admission the patient’s hemoglobin was 7.8 g/dL, the hematocrit was 26%, platelets were 346,000/mm3, prothrombin time (ET) was 12 seconds, partial thromboplastin time (ETT) was 30 seconds, fibrinogen was 335 mg%, fibrin split products were not detectable, and the ristocetin factor VIII cofactor was 1.13. All coagulation factors appeared normal except for the Ivy bleeding time, which was at the high range of normal at 9.5 minutes (range, 2.5 to 9.5 minutes). The patient had not been on any nonsteroidal anti-inflammatory drugs, and her family history and past history were negative for bleeding problems. The hematologist who saw her in consultation did not feel her bleeding was secondary to a coagulopathy. He suggested she be given 2 U of packed red blood cells and advised placing her on prophylactic Amicar (aminocaproic acid; Lederle, Markham, Ontario, Canada) 4 g every 4 hours for 7 days. No further bleeding episodes occurred until 11 days later, which was 11 weeks following her original orthognathic surgery. The patient on awakening in the morning had noticed some swelling in the right cheek and subsequently had a small amount of bleeding from the mouth and nose, which stopped spontaneously. An hour later, however, more vigorous bleeding commenced, although this was stopped temporarily with two squirts of Afrin (oxymetazoline hydrochloride; Schering, Kenilworth, NJ) nasal spray into her right nostril. The patient was readmitted to hospital as she continued to experience some epistaxis. She underwent a right carotid arteriogram that revealed a 9 x 13-mm vascular malformation that was apparently a pseudoaneurysm of the sphenopalatine branch of the maxillary artery. Selective embolization of this malformation could not be carried out, however, because catheterization of the distal aspect of the maxillary artery could not be accomplished because of spasm of the maxillary artery. The patient was actively bleeding at the time of the arteriogram. Because of poor control of the bleeding it was decided to take the patient back to the operating room. In conjunction with an ENT surgeon, a transantral approach to ligate the right maxillary artery was attempted. This was unsuccessful because of altered anatomy and marked hemorrhage which resulted in poor visualization. The right external carotid artery was therefore ligated in the neck above the lingual artery. This signit’icantly decreased the bleeding, which was then stopped by additional packing in the posteriomedial aspect of the maxilla with strips of Surgicel. The patient received an additional 2 U of whole blood to increase her hematocrit to 34% at
SURGERY-FALSE
ANEURYSMS
AND A-V FISTULAS
the time of discharge. She was placed on prophylactic Amicar 4 g every 4 hours for 2 weeks at the suggestion of the hematologist. No other active treatment was carried out at that time other than a continuation of her limitation of physical activity. The neuroradiologist was consulted about the feasibility of selective embolization of the contralateral maxillary artery due to concerns about revascularization of the right maxillary artery occurring from the left side. The neuroradiologist advised against this because he felt there was a possibility of dislodging the clot from the right maxillary artery during the arteriograms. The patient fortunately had no further bleeding episodes. Case 3 A 29-year-old man with maxillary retrusion secondary to a previous Le Fort II fracture in a motor vehicle accident 2 years previously was treated with a Le Fort I maxillary osteotomy with advancement. Slight nonspecific bleeding was noted from the left posterior maxillary region at the time of surgery. The estimated blood loss at surgery was 1,000 mL and the patient received 1 U of autologous packed red blood cells intraoperatively. His initial course was uneventful until 6 days postoperatively when, at approximately 5:OOPM, he developed intractable bleeding from his nose and mouth, especially the left nostril. The bleeding had been brought on when he blew his nose following exercising by lifting weights. The patient was taken to the emergency department at a local hospital where it took 4 hours to bring the bleeding under control. The bleeding slowed significantly after an ENT surgeon placed left anterior and posterior nasal gauze packs and a balloon catheter as a right posterior nasal pack. The nose bleed was persistent through the evening, but by early the next morning the epistaxis had significantly decreased and the patient was considered stable; no bleeding was noted going back down into the pharynx. On the second day after the bleeding had commenced the patient was transfused with 4 U of packed red blood cells because of his low hemoglobin. The nasal packs were left in place for a total of 5 days, and the patient was hospitalized for 1 week in total. Two days following his discharge he was admitted to a larger medical center for arteriograms. The patient was not experiencing any active bleeding at that time. A neuroradiologist performed arteriography of the left and right external carotid artery systems. A small false aneurysm of the left maxillary artery was noted. The neuroradiologist embolized both the left and right maxillary and facial arteries in an attempt to prevent any recurrence of the epistaxis. The patient was discharged from hospital the following day. The rest of the patient’s postoperative course was uneventful, with no long-term sequelae.
Case 4 A 27-year-old woman with a midfacial retrusion secondary to a left unilateral cleft lip and palate deformity underwent a Le Fort I osteotomy with advancement and a bone graft to the alveolar cleft site. The surgeon encountered difficulty in downfracturing the maxilla due to very thick bone, especially along the posterior maxillary walls. Excessive generalized bleeding was noted intraoperatively, but no specific vessel could be identified as the source of the hemorrhage. The patient received 3 U of packed red blood cells intraoperatively. Postoperatively,
LANIGAN
575
ET AL
in the recovery room, the patient was noted to have chemosis of the left eye, mild left ptosis, diplopia, and an inability to move the left eye laterally from the midline due to a left VI nerve palsy. A computed tomography (CT) scan was reported as normal, but a left carotid arteriogram demonstrated an extensive left carotidcavernous fistula. The patient was referred to another institution where successful balloon embolization of the carotid-cavernous fistula was carried out. The patient’s double vision gradually resolved as the VI nerve palsy improved. By 6 months postoperatively her vision was normal, the ptosis had resolved, and the “pulsating pressure” that she once had in the temporal region had disappeared. The patient, however, continued to complain of mild pain in the maxillary region bilaterally.
Discussion From a review of the literature, and from the cases presented in this report, it is possible to draw several conclusions regarding false aneurysms and A-V listulas following orthognathic surgery. False aneurysms following mandibular orthognathic surgery, especially vertical or oblique mandibular ra-
mus osteotomies, are most likely to involve the internal maxillary artery,’ which is usually lacerated when the osteotomy cut is made near the sigmoid notch.26 Care must be taken to avoid saws that cut so effectively that they not only section the bone well, but also cut through into the soft tissues along the medial side of the ascending ramus. Marked bleeding is usually encountered at the time of surgery, which may be satisfactorily controlled intraoperatively by pressure packing. Signs of a false aneurysm, such as an enlarging pulsatile mass or recurrent bleeding, may eventually become obvious. The vessel most commonly involved with false aneurysms following maxillary orthognathic surgery is the internal maxillary artery, in particular the sphenopalatine branch. The internal maxillary artery and its branches are most vulnerable to damage in their location in the pterygopalatine fossa when the maxillary tuberosity is separated from the pterygoid plates with an osteotome27~28 or during the downfracture procedure.29 The classical signs of a false aneurysm, such as a bruit or palsatile expanding mass, may not be evident from vessels damaged during a Le Fort I osteotomy due to their deep location. The first indication of the problem may be persistent or recurrent epistaxis that does not respond to measures such as anterior and posterior nasal packing. One factor that may be unusual in their presentation is the time at which the initial bleeding first occurs. In the series of cases of hemorrhage following Le Fort I osteotomies previously reported, 29.30the vast majority of the initial episodes occurred within the first 14 days postoperatively. The initial bleeding associated with a false aneurysm can present later than this, how-
ever. The patient in case 1 experienced her first episode of significant epistaxis 6 weeks postoperatively, whereas the patient in case 2 had her initial episode of bleeding 3 weeks after surgery. The patient reported by Solomons and Blumga& first presented with epistaxis 1 month postoperatively. Ideally, any patient who presents with the first episode of epistaxis after the second postoperative week, or who experiences recurrent significant hemorrhage following orthognathic surgery, should be investigated by angiography to rule out a false aneurysm as a source of the hemorrhage. If the patient is actively bleeding at the time of the investigation, angiography may be able to locate the actual site of the hemorrhage. Angiography also helps in delineating the development of collateral blood flow to an area, and whether or not the contralateral arterial tree is contributing to the problem.3’ If a false aneurysm is diagnosed during angiography, the treatment of choice is transcatheter embolization.“,6 Surgery can also be used to resect the false aneurysm (see case l), but this treatment should only be used if it can be done safely without placing adjacent structures at risk, or risking rupture of the false aneurysm.” Schwartz reported a case” of a false aneurysm of the internal maxillary artery following Le Fort II and mandibular fractures. Embolization failed to control the aneurysm and its size and location made a direct surgical approach to resect it unsafe. Surgery was therefore performed to clip the third portion of the internal maxillary artery via a transantral approach, as well as to ligate the distal branches of the external carotid artery that might provide collateral flow to the lesion, including the common facial-lingual trunk, and the ascending pharyngeal, occipital, and superficial temporal arteries. One of the advantages of embolization over surgical intervention is that the more distal vessels that supply the bleeding source may be obliterated while sparing the more proximal vessels.3’ This is particularly important following orthognathic surgery where one wishes to avoid a further compromise of an already diminished vascular supply, which could contribute to the development of aseptic necrosis of the maxilla.33 Selective embolization as distally as possible will also minimize the risk of persistent hemorrhage from collateral flow. With false aneurysms, embolization with agents such as small pieces of Gelfoam may be unsuccessful and devices such as Gianturco coils may have to be used.2,6*8 Arteriovenous fistulas following orthognathic surgery are more apt to involve large vessels, especially the internal carotid artery.3-5’7 The basis for injuries to the internal carotid artery following orthognathic surgery has been discussed by Lani-
576
ORTHOGNATHIC
gan.25 Direct trauma to the artery can occur during the pterygomaxillary dysjunction or the maxillary downfracture when a basal skull fracture ensues that involves areas such as the foramen lacerum or the carotid canal. Indirect trauma can follow sudden forceful stretching of the internal carotid artery in the neck over the upper cervical spine by cervical hyperextension with contralateral rotation of the head. A severe disruption of the layers of the wall of the internal carotid artery could lead to the formation of an A-V listula. A carotid-cavernous fistula may be clinically obvious early in the postoperative course,7 or the development of signs and symptoms may be delayed for weeks or months.3*4 The diagnosis can be confirmed by carotid angiography. In all the cases of carotid-cavernous sinus fistula following orthognathic surgery reported to date3,4*7 (case 4), the tistulas have been successfully obliterated by detachable balloon catheters, with no recurrence. Matching the size of the embolic material to the shunt is critical for success with this procedure. The advantage of the balloon catheter technique over techniques used in the past is that it can obliterate the tistula more safely, while preserving blood flow in the internal carotid artery.4*7 Because embolization may not totally seal the shunt in an A-V tistula, there is always the risk that the defect may gradually reopen. 24 Embolization may be repeated, if necessary, however. The occurrence of direct trauma to the carotid artery secondary to basal skull fractures would appear to be related to difficult downfractures of the maxilla4 (case 4). If a difftcult downfracture is encountered following completion of routine Le Fort I osteotomies, then the posterior walls of the maxilla should be sectioned with an osteotome or, alternatively, sectioning to achieve the pterygomaxillary dysjunction can be attempted through the maxillary tuberosity rather than between the tuberosity and pterygoid plates34 in an attempt to avoid, as much as possible, the thick posterior maxillary walls. If a relatively atraumatic downfracture still cannot be achieved following these maneuvers, consideration should be given to stopping the procedure and obtaining a CT scan to look at the posterior maxilla/pterygoid plate regions. If the CT scan shows excessive thickness of bone in this region, or abnormalities in the base of the skull that could increase the relative risk of an injury to the internal carotid artery system or of hemorrhage from damage to the maxillary artery or its branches, then the surgeon may wish to discuss these possibilities further with the patient prior to proceeding with additional surgery. In certain patients with craniofacial malformations it may be prudent to do the
SURGERY-FALSE
ANEURYSMS
AND A-V FISTULAS
CT scan even before proceeding with the initial surgery. 35 Surgeons should also be aware of the possibility that congenital A-V malformations that could lead to marked intraoperative and/or postoperative hemorrhage might be encountered while performing orthognathic surgery. Lanigan et al*‘j reported a case of major intraoperative bleeding from a congenital A-V malformation that was lacerated along the medial surface of the mandible when an intraoral vertical mandibular ramus osteotomy was performed for unilateral condylar hyperplasia. Acknowledgment The authors would like to thank the surgeons who responded to our inquires, often on more than one occasion, for details regarding their specific cases. We would also like to thank the patient in case 3 who also responded to our inquiry with additional information regarding his complication.
References I. Clark R, Lew D, Giyanani VL, et al: False aneurysm com2.
3. 4.
5.
6.
7.
8.
9. 10.
11.
12. 13. 14. 15.
16.
plicating orthognathic surgery. J Oral Maxillofac Surg 4557, 1987 Hemmig S, Johnson RS, Ferraro N: Management of a ruptured pseudoaneurysm of the sphenopalatine artery following a Le Fort I osteotomy. J Oral Maxillofac Surg 45533, 1987 Habal M: A carotid cavernous sinus fistula after maxillary osteotomy. Plast Reconstr Surg 77:981, 1986 Lanigan D, Tubman D: Carotid-cavernous sinus fistula following orthognathic surgery. J Oral Maxillofac Surg 45:%9, 1987 Newhouse R, Schow S, Kraut R, et al: Life-threatening hemorrhage from a Le Fort I osteotomy. J Oral Maxillofat Surg 40: 117, 1982 Solomons NB, Blumgart R: Severe late-onset epistaxis following Le Fort I osteotomy: Angiographic localization and embolization. J Laryngol Otol 102:260, 1988 Hes J, de Man K: Carotid-cavernous sinus Iistula following maxillofacial trauma and orthognathic surgery. Int J Oral Maxillofac Surg 17:295, 1988 Lustbader D, Schwartz M, Zito J, Stem M: The use of percutaneous transcatheter embolization to control postoperative bleeding following Le Fort I osteotomy: Report of three cases. J Oral Maxillofac Surg (accepted) D’Orta J, Shatney CH: Post-traumatic pseudoaneurysm of the internal maxillary artery. J Trauma 22: 161, 1982 Field PL, Tress BM, O’Brien MD, et al: Embolization of traumatic aneurysm of the maxillary artery. Aust NZ Surg 49:244, 1979 Callem WS: Traumatic (false) aneurysms of the terminal portion of the external carotid artery. Am J Surg 106:522, 1963 Bresner M, Brekke J, Dubit J, et al: False aneurysms of the facial region. J Oral Surg 30:307, 1972 Wineland PL. Topazian RG, Marble HB: False aneurysm of the facial artery. J Oral Surg 34642, 1976 Tatelman M: Arteriography in oral and maxilofacial surgery. J Oral Surg 34:795, 1976 Marks MW, Argenta LC, Dingman RO: Traumatic arteriovenous malformation of the external carotid arterial system. Head Neck Surg 6:1054, 1984 Guzman Freire JE, Cabanas Valls R, Munoz Noya JA, et al: Arteriovenous fistulas of the anterior facial vessels: Review of the literature and report of case. J Oral Surg 32: 150, 1974
LANIGAN
577
ET AL
17. Cooperband B. Friedel W, Bhatt GM, et al: False aneurysm of the facial artery. J Oral Maxillofac Surg 47: 1327, 1989 18. Taylor DV: Traumatic aneurysm and facial palsy as a complication of a mandibular fracture. Br J Oral Surg 4:202, I%7 19. Schwartz H. Kendrick R, Pogorel B: False aneurysm of the maxillary artery. An unusual complication of closed facial trauma. Arch Otolaryngol 109:616, 1983 20. Van den Akker HP, Van der Lijn FA: A false aneurysm of the facial artery as a complication of circumferential wiring. Oral Surg 37514, 1974 21. Miller FJ, Entzminger LB, Coleman LL, et al: Successful catheter embolization of a false aneurysm of the internal maxillary artery. Arch Otolaryngol 101:517, 1975 22. Peoples JR, Herbosa EG, Dion J: Management of internal maxillary artery hemorrhage from temporomandibular joint surgery via selective embolization. J Oral Maxillofac Surg 46: 1005, 1988 23. Bradley P: Conservative treatment for temporomandibular joint pain dysfunction. Br J Oral Maxillofac Surg 25: 125. 1987 24. Brown JS, Ward-Booth RP: Arterio-venous malformation following a radical neck dissection. Int J Oral Maxillofac Surg 18:239, 1989 25. Lanigan DT: Injuries to the internal carotid artery following orthognathic surgery. Int J Adult Ortho Orthognath Surg 4:215. 1988 26. Lanigan DT. Hey JH. West RA: Hemorrhage following man-
dibular osteotomies. A report of twenty-one cases. J Oral Maxillofac Surg (accepted) 27. Turvey T, Fonseca R: The anatomy of the maxillary artery in the pterygopalatine fossa: Its relationship to maxillary surgery. J Oral Surg 38:92, 1980 28. Robinson P, Hendy C: Pterygoid plate fractures caused by the Le Fort I osteotomy. Br J Oral Maxillofac Surg 24:198, 1986 29. Lanigan DT, Hey JH, West RA: Major vascular complications of orthognathic surgery: Hemorrhage associated with Le Fort I osteotomies. J Oral Maxillofac Surg 48:561.
1990 30. Lanigan DT, West RA: Management of postoperative hemorrhage following the Le Fort I maxillary osteotomy. J Oral Maxillofac Surg 42:367, 1984 3 1. Schilstra S, Marsman J: Embolization for traumatic epistaxis. J Craniomaxillofac Sure. 15:28. 1987 32. Kingsley D, O’Conner A: Embolization in otolaryngology. J Laryngol Otol 96:439, 1982 33. Lanigan DT. Hey JH. West RA: Aseptic necrosis of the maxilla. Report of 36 cases. J Oral Maxillofac Surg 48:142, 1990 34. Timble LD. Tideman H, Stoelinga P: A modification of the pterygoid plate separation in low-level maxillary osteotomies. J Oral Maxillofac Surg 41:544, 1983 35. Reaume C, MacNichol B: Complications encountered during Le Fort I osteotomy in a patient with mandibulofacial dysostosis. J Oral Maxillofac Surg 46:1003. 1988
