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Massive epistaxis due to pseudoaneurysm of the sphenopalatine artery: a rare post-operative complication of orthognathic surgery

Published online by Cambridge University Press:  22 April 2013

Y-W Kim ,
M-J Baek ,
H-D Kim  and
K-S Cho
Y-W Kim
Affiliation:
Department of Otorhinolaryngology, Inje University College of Medicine, Haeundae Paik Hospital, Busan, South Korea
M-J Baek
Affiliation:
Department of Otorhinolaryngology, Inje University College of Medicine, Haeundae Paik Hospital, Busan, South Korea
H-D Kim
Affiliation:
Department of Radiology, Inje University College of Medicine, Haeundae Paik Hospital, Busan, South Korea
K-S Cho*
Affiliation:
Department of Otorhinolaryngology and Biomedical Research Institute, Pusan National University School of Medicine, Busan, South Korea
*
Address for correspondence: Dr K-S Cho, Department of Otorhinolaryngology and Biomedical Research Institute, Pusan National University School of Medicine, 1-10 Ami-dong, Seo-gu, Busan 602-739, South Korea Fax: 82-51-246-8668 E-mail: choks@pusan.ac.kr
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Abstract

Objective:

To introduce pseudoaneurysm of the sphenopalatine artery as the possible aetiology of acute massive epistaxis in patients with a history of orthognathic surgery accompanied by Le Fort I osteotomy.

Methods:

Case report and literature review.

Results:

This paper reports a case of acute life-threatening epistaxis following Le Fort I osteotomy. Computed tomography and angiography showed a pseudoaneurysm of the sphenopalatine artery, which was successfully treated by endovascular embolisation.

Conclusion:

Although a pseudoaneurysm of the sphenopalatine artery following Le Fort I osteotomy is extremely rare, it should be considered as the possible aetiology of acute massive epistaxis in patients with a history of orthognathic surgery accompanied by Le Fort I osteotomy.

Keywords

EpistaxisAneurysmFalsePostoperative ComplicationsOrthognathic SurgeryOsteotomyLe Fort

Information

Type
Clinical Records
Information
The Journal of Laryngology & Otology , Volume 127 , Issue 6 , June 2013 , pp. 610 - 613
Copyright
Copyright © JLO (1984) Limited 2013 

Introduction

Although Le Fort I osteotomy is one of the most commonly performed surgical procedures for the correction of dental and facial deformities in orthognathic surgery,Reference Chepla, Totonchi, Hsu and Gosain1–Reference Panula, Finne and Oikarinen2 post-operative complications from vascular injuries are rare.Reference Panula, Finne and Oikarinen2 Arterial pseudoaneurysm resulting from blunt or, more commonly, sharp trauma to the vessel wall has been reported,Reference Chepla, Totonchi, Hsu and Gosain1–Reference Zachariades, Rallis, Papademetriou, Papakosta, Spanomichos and Souelem3 but pseudoaneurysm of the sphenopalatine artery following Le Fort I osteotomy is extremely rare. We report a case of acute, massive, life-threatening epistaxis due to a pseudoaneurysm of the sphenopalatine artery as a post-operative complication of orthognathic surgery.

Case report

A 31-year-old man underwent Le Fort I osteotomy with bilateral sagittal split ramus osteotomy of maxillar advancement and mandibular setback for maxillar and mandibular deformities in a local dental clinic. Two weeks later, he presented with a massive epistaxis of both sides and oral bleeding.

Upon physical examination, he was pale, shivering and in a confused state. Nasal packing was performed using Merocel® packs. Complete blood count results demonstrated haemoglobin levels of 7.6 g/dl, haematocrit of 18.8 per cent and platelet count of 475 × 103/μl. Computed tomography (CT) with contrast showed a nodular, vascular, enhancing lesion, measuring 1.2 cm at the posterior aspect of the right maxillary sinus fed by the right internal maxillary artery, which was suggestive of a pseudoaneurysm or other vascular malformation (Figure 1).

Fig. 1

Pre-operative axial (a) and coronal (b) computed tomography images, which show a nodular vascular enhancing lesion (arrow) measuring 1.2 cm at the posterior aspect of the right maxillary sinus, suggestive of pseudoaneurysm or other vascular malformation.

The patient was sent to interventional radiology for angiography and embolisation. Angiography revealed a large 2-cm pseudoaneurysm of the right sphenopalatine artery without collateral circulation between the internal and external carotid branches (Figure 2). Superselective embolisation with lipiodol and N-butyl-2-cyanoacrylate was performed.

Fig. 2

Sagittal (a) and coronal (b) superselective angiographic images, which show a central collection of contrast, indicating a 2-cm pseudoaneurysm of the right sphenopalatine artery.

The post-embolisation angiogram showed complete resolution of the pseudoaneurysm (Figure 3). A total of two units of packed red cells and three units of fresh frozen plasma were required to stabilise the patient. He remained asymptomatic during the six-month follow up, with no further episodes of bleeding.

Fig. 3

Sagittal (a) and coronal (b) post-embolisation angiographic images showing total obliteration of the pseudoaneurysm, which was achieved by infusion of N-butyl-2-cyanoacrylate into the affected artery and pseudoaneurysm.

Discussion

The Le Fort I osteotomy involves separating the maxilla and the palate from the skull above the roots of the upper teeth.Reference Chepla, Totonchi, Hsu and Gosain1 The operation is frequently carried out with mandibular sagittal split osteotomy for the correction of dentofacial deformity. Osteotome positioning and manipulation are commonly used in procedures in order to mobilise and reposition the mandible and maxilla. The proximity of the osteotome to the internal maxillary artery and branches could increase the potential for injury at this junction.Reference Chepla, Totonchi, Hsu and Gosain1 However, pseudoaneurysm formation from the sphenopalatine artery is extremely rare because its diminutive calibre and deep location in the mid-face have proven protective from blunt and superficially penetrating injuries.Reference Zachariades, Rallis, Papademetriou, Papakosta, Spanomichos and Souelem3

Arterial pseudoaneurysm can occur as a result of blunt or, more commonly, sharp trauma to the vessel wall.Reference Bradley, Elahi and Kawamoto4 A pseudoaneurysm is an abnormal dilatation of an artery resulting from incomplete disruption of the arterial wall, which leads to an expanding lesion between the damaged artery and the surrounding tissues. After disruption of the arterial wall, haemorrhage occurs in the partially transected vessel into the adjacent tissues until the pressure of the formed haematoma counterbalances the arterial pressure, which compresses the defect and stabilises the bleeding.Reference Silva, O'Ryan, Beckley, Young and Poor5 The rarity of a pseudoaneurysm in the external carotid artery is due to the small size of the branches of the external carotid artery, which makes complete cross-cutting more likely than partial laceration. Moreover, larger calibre branches are protected by thicker tissue.Reference Manafi, Ghenaati, Dezham and Arshad6 Most cases of pseudoaneurysm of the external carotid artery have been reported to appear in the superficial temporal artery and facial artery. These branches are more vulnerable because of their large vessel diameter, long course and relatively superficial position.Reference Zachariades, Rallis, Papademetriou, Papakosta, Spanomichos and Souelem3, Reference Pappa, Richardson and Niven7 Our case showed acute massive life-threatening epistaxis due to a pseudoaneurysm of the sphenopalatine artery following orthognathic surgery, which was successfully treated by endovascular embolisation.

The diagnosis of a pseudoaneurysm is often clinical, and this is then confirmed by radiological studies. Although the clinical findings of a pulsatile mass with pain and an audible thrill are highly diagnostic, these common findings are not found in a pseudoaneurysm of the sphenopalatine artery because of the deep location of the artery. The only presentation may be mild to severe oropharyngeal bleeding and epistaxis, as in our case. Computed tomography is recommended as a diagnostic tool because of its capacity to reveal anatomical details of vascular injuries with high definition. However, CT may not allow direct visualisation of a pseudoaneurysm of the sphenopalatine artery, especially if it is small or obscured by a haematoma or oronasal bleeding. Angiography (a radiological technique) can reveal the pathways feeding an injury and enable localisation of the exact site of the bleeding, thereby allowing precise treatment. Therefore, angiography is the diagnostic examination of choice when investigating the major cause of bleeding following a Le Fort I osteotomy.Reference Bynoe, Kerwin, Parker, Nottingham, Bell and Yost8 In the present case, a selective angiogram of the right external carotid artery was performed, which revealed a large pseudoaneurysm irrigated by the sphenopalatine artery.

  • • Delayed life-threatening epistaxis due to pseudoaneurysm of the sphenopalatine artery is a rare complication following Le Fort I osteotomy

  • • However, otolaryngologists should be aware of this potential complication and treatment options

  • • This case report describes an episode of massive epistaxis that occurred two weeks after a Le Fort I osteotomy

  • • Angiography revealed a pseudoaneurysm of the sphenopalatine artery, successfully treated by endovascular embolisation with N-butyl-2-cyanoacrylate

  • • High clinical suspicion for pseudoaneurysm should be maintained in patients with delayed post-operative bleeding following orthognathic surgery

Most aneurysms of the face may be treated with vascular ligation or direct repair. However, surgical approaches to a pseudoaneurysm of the sphenopalatine artery can be difficult due to deep location, especially in massive life-threatening epistaxis. Angiographic embolisation has been shown to be safe and effective for the treatment of head and neck haemorrhage.Reference Bynoe, Kerwin, Parker, Nottingham, Bell and Yost8 The use of newer techniques and materials in angioembolisation has further improved success rates and reduced the morbidity associated with surgical exploration. Of the numerous embolising materials, N-butyl-2-cyanoacrylate has the advantages of good penetration and free dispersion in comparison with other materials of embolisation, and provides rapid, permanent induction of thrombosis and occlusion of the pseudoaneurym.Reference Zhang, Xie, You, Mao, Wang and He9 In the present case, superselective endovascular embolisation with N-butyl-2-cyanoacrylate was performed successfully, and the patient remained asymptomatic after more than six months follow up.

References

1Chepla, KJ, Totonchi, A, Hsu, DP, Gosain, AK. Maxillary artery pseudoaneurysm after Le Fort I osteotomy: treatment using transcatheter arterial embolization. J Craniofac Surg 2010;21:1079–81CrossRefGoogle Scholar
2Panula, K, Finne, K, Oikarinen, K. Incidence of complications and problems related to orthognathic surgery: a review of 655 patients. J Oral Maxillofac Surg 2001;59:1128–36CrossRefGoogle ScholarPubMed
3Zachariades, N, Rallis, G, Papademetriou, G, Papakosta, V, Spanomichos, G, Souelem, M. Embolization for the treatment of pseudoaneurysm and transection of facial vessels. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:491–4CrossRefGoogle ScholarPubMed
4Bradley, JP, Elahi, M, Kawamoto, HK. Delayed presentation of pseudoaneurysm after Le Fort I osteotomy. J Craniofac Surg 2002;13:746–50CrossRefGoogle ScholarPubMed
5Silva, AC, O'Ryan, F, Beckley, ML, Young, HY, Poor, D. Pseudoaneurysm of a branch of the maxillary artery following mandibular sagittal split ramus osteotomy: case report and review of the literature. J Oral Maxillofac Surg 2007;65:1807–16CrossRefGoogle ScholarPubMed
6Manafi, A, Ghenaati, H, Dezham, F, Arshad, M. Massive repeated nose bleeding after bimaxillary osteotomy. J Craniofac Surg 2007;18:1491–3CrossRefGoogle ScholarPubMed
7Pappa, H, Richardson, D, Niven, S. False aneurysm of the facial artery as complication of sagittal split osteotomy. J Craniomaxillofac Surg 2008;36:180–2CrossRefGoogle ScholarPubMed
8Bynoe, RP, Kerwin, AJ, Parker, HH 3rd, Nottingham, JM, Bell, RM, Yost, MJ et al. Maxillofacial injuries and life-threatening hemorrhage: treatment with transcatheter arterial embolization. J Trauma 2003;55:74–9CrossRefGoogle ScholarPubMed
9Zhang, C, Xie, X, You, C, Mao, B, Wang, C, He, M et al. Endovascular treatment of traumatic pseudoaneurysm presenting as intractable epistaxis. Korean J Radiol 2010;11:603–11CrossRefGoogle ScholarPubMed
Figure 0

Fig. 1 Pre-operative axial (a) and coronal (b) computed tomography images, which show a nodular vascular enhancing lesion (arrow) measuring 1.2 cm at the posterior aspect of the right maxillary sinus, suggestive of pseudoaneurysm or other vascular malformation.

Figure 1

Fig. 2 Sagittal (a) and coronal (b) superselective angiographic images, which show a central collection of contrast, indicating a 2-cm pseudoaneurysm of the right sphenopalatine artery.

Figure 2

Fig. 3 Sagittal (a) and coronal (b) post-embolisation angiographic images showing total obliteration of the pseudoaneurysm, which was achieved by infusion of N-butyl-2-cyanoacrylate into the affected artery and pseudoaneurysm.