“Signs and Symptoms Tell All”–Pseudoaneurysm as a Cause of Postoperative Bleeding after Orthognathic Surgery–Report of a Case and a Systematic Review of Literature

Abstract

Purpose

Pseudoaneurysms are one of the rare complications that can be encountered after the orthognathic surgery. We are presenting a new case of pseudoaneurysm of bilateral sphenopalatine artery after Bijaw Surgery in a young male and a systematic review of all the cases in the literature emphasizing on signs and symptoms, epistaxis or bleeding episodes and treatment outcomes.

Methods

A systematic research strategy was planned according to the PRISMA guidelines, and articles were taken from 1986 to September, 2019. A total of 899 articles were selected for screening, out of which only 26 articles met our inclusion and exclusion criteria. These were included in the study for qualitative analysis.

Results

Most PAs were associated with Lefort I osteotomy (69.7%), followed by sagittal split osteotomy (24.24%). Average intraoperative blood was 635 ml. Maximum number of episodes of epistaxis/swelling or bleeding occurred in second week. Mean bleeding episodes were 2.58 ± 0.996. The arteries commonly affected were internal maxillary artery (42%), sphenopalatine artery (27.27%), facial artery (15.15%), descending palatine artery (12.12%), internal carotid artery (9.09%) and infraorbital artery (3.03%). Embolization was treatment of choice in 81.81% cases.

Conclusion

If a patient has recurrent epistaxis or swelling after orthognathic surgery, it is advisable to go for diagnostic imaging like angiography without any delay. In recent times, advanced techniques and expertise are readily available for early diagnosis and management of pseudoaneurysm.

Introduction

Orthognathic procedures like Lefort I osteotomy and bilateral sagittal split osteotomy (BSSO) are frequently performed for the surgical correction of the dentofacial abnormalities. These procedures are relatively safe, but inevitably there may be undesirable intraoperative and postoperative complications.

The most serious and life-threatening among these complications are vascular injuries which include hemorrhage, thromobosis, arteriovenous fistulae and pseudoaneurysms [1, 2]. False aneurysm or pseudoaneurysm (PA) is abnormal dilatation of an artery resulting from breach in the vessel wall, mainly due to trauma. This leads to leakage of the blood through the wall forming an irregular, well-contained lesion; the vessel lumen and aneurysm form a direct communication of blood flow through a hole in the vessel wall. Whereas a true aneurysm is the one that involves all the three layers of the wall of the artery, i.e., intima, media and adventitia, it is an abnormal dilatation of an intact artery or vein without any history of trauma. It is thick walled with a smooth outline and has less chances of rupture. PA has higher chances of rupture than a true aneurysm of comparable size mainly due to poor wall support.

The pathophysiology for the development of a PA is multifactorial including trauma, iatrogenic causes, infection, inflammation or tumor, although trauma remains the most common causative factor [3]. The most prevalent age for a traumatic aneurysm to occur is between 20 and 40 years [4]. Three factors that determine the development of a PA are magnitude of vascular injury, rate of blood flow in the vessel and elasticity of the surrounding structures. Platelet occlusion becomes difficult if the magnitude of injury is more or the arterial blood flow is high. Elasticity of the neighboring fibrous tissue plays a major role as thick tissue usually counteracts the bleeding, leading to hematoma formation but loose fragile tissue will lead to a persistent hemorrhage [3]. This hematoma can either resolve on its own or there might be an initiation of the formation of pseudoaneurysm. If the blood flow leakage between the hematoma and laceration is maintained, it leads to liquefaction of the center. The perivascular tissue form a sac lined with tissue that resemble endothelial cells in inner layer and outer fibrous capsule. The arterial pressure leads to gradual expansion and may eventually increase in size or rupture resulting in massive bleeding as described in Fig. 1.

Fig. 1.

Flowchart for pathophysiology of PA

PAs of the internal maxillary artery(IMA), sphenopalatine artery, descending palatine artery and facial artery have been reported in the literature after orthognathic surgery, which can be life-threatening [5–9] Awareness of the signs and symptoms and potential treatment outcomes by the cranio/oral maxillofacial surgeons is very important to prevent any untoward consequences. The purpose of this research was to do a systematic review of all the cases in literature with pseudoaneurysm after orthognathic surgery and to report a new case.

Case Report

A young male aged 21 years underwent maxillary impaction of 7 mm at Lefort I level and mandibular advancement by 4 mm with BSSO with correction of gummy smile. Extraction of the third molars was done 6 months prior to surgery with delayed healing of extraction socket. Preoperative hemoglobin was 12.5 g/dl with a normal hemogram and coagulogram except for a mildly raised aPTT (44 s). Surgery was performed uneventfully with an intraoperative blood loss of around 450 ml. Hemostasis was achieved and primary closure done.

On the first postoperative day (POD 1), about 3 ml of hematoma was drained from right maxillary anterior vestibular region. Patient was discharged on POD 4 with mild postoperative edema of face and no recurrence of bleeding or discharge. Then, on POD 9, patient noticed bleeding from right nostril for which nasal pack and Injection Tranexamic Acid 500 mg was administered at the local hospital and patient was referred back to our department for further management. On POD 10, there was mild bleeding from nose along with a difficulty in respiration due to the blocked nose. Endoscopy was performed immediately wherein a clot was detected in posterior nasal cavity, which could not be removed. CT angiography (CTA) was advised to locate the bleeder and the radiologist confirmed the presence of a small pseudoaneurysm in right maxillary artery (distal branch of internal maxillary artery) which was embolized through femoral artery percutaneous embolization with gel foam under local anesthesia (Fig. 2 a, b). Bleeding stopped postprocedure but nasal blockage persisted. On POD 15, patient was discharged under hemodynamically stable conditions.

Fig. 2.

a,b Digital subtraction angiography (Lateral view) demonstrating the pseudoaneurysm of right sphenopalatine artery (a) (arrow) and after embolization (b)

After two days, there was fresh episode of epistaxis and Hb decreased to 7 g/dl. The patient was readmitted and CTA was repeated. Fresh CTA suggested that the contrast filled a midline outpouching measuring 5 × 4 × 3.9 mm in size. This PA was seen in the midline, slightly in a left paramedian location within the nasal cavity, just above the posterior aspect of hard palate, likely arising from distal branch of sphenopalatine artery (Fig. 3 a, b, c).

Fig. 3.

a,b,c A small pseudoaneurysm in left paramedian nasal cavity CT angiography in axial, sagittal and coronal sections which was missing in first CTA

This was a new finding and was not appreciated on the previous CTA. Tiny PA found in the previous CTA was not filling on the fresh CTA, both in the arterial and venous phase images. Embolization was repeated (Fig. 4a,b) with an uneventful postoperative course.

Fig. 4.

a,b Complete resolution of PA after embolization

Patient was kept under observation for three more days with transfusion of 4 units of Fresh frozen plasma along with 2 units of PRBCs in view of previous delayed healing of surgical wounds and increased APTT than normal, in consultation with the hematologist. Factor 8 assay was performed to rule out coagulopathy which was found to be slightly less than normal range 54% (60–100%) but was considered normal for O positive individuals. No reoccurrence of bleeding was reported, even after 1 year of follow-up.

Research Strategy

The aim of the literature search was to identify and enlist the worldwide reported cases of pseudoaneurysms after orthognathic surgery so as to understand the age and gender predilection, vessel involved, intraoperative blood loss, time of first bleeding event after the surgery, number of bleeding events and its management.

Materials and Methods

Two researchers individually did systematic search on PubMed and Ovid using terms “pseudoaneurysm”, “false aneurysm”, “bleeding”, “orthognathic”, “lefort*’, “BSSO”, “Bilateral sagittal split osteotomy” and ”osteotomy”. We thoroughly cross checked the references of these articles and manual search was performed on Google scholar. Final selection of the articles was made based on the inclusion and exclusion criteria.

Inclusion Criteria

1. Cases causing postoperative bleeding after orthognathic surgery due to pseudoaneurysm.

2. Details of cases available.

3. English language/translated articles only.

4. From the year 1986 to September 2019.

Exclusion Criteria

1. Incomplete text available.

2. Other causes of pseudoaneurysms like trauma.

Results

Paper Selection

A total of 883, 18, 6 articles were identified via Pubmed, Ovid and manual search, respectively. After duplication removal, 889 articles were selected for screening. Of these, only 26 articles met the exclusion and inclusion criteria which were included in the study for qualitative analysis.

1. Age

Mean age 25.06 ± 7.48 years with minimum age of 13 years to maximum age of 42 years.

• (b)Gender

A total of 33 patients in 27 studies were enrolled for our review. Out of 33 patients, 9 were females and 24 being males.

• (c)Procedure

Most PAs were associated with Lefort I osteotomy (69.7%), followed by sagittal split osteotomy (24.24%) and other surgical procedures (6.06%) as described in Fig. 5.

• (d)Intraoperative blood loss

Fig. 5.

Pie chart depicting procedures associated with PAs, Lefort I osteotomy being most commonly responsible

There were 10 patients (N = 10) who had values mentioned for the intraoperative blood loss. Average blood loss was about 635 ml.

• (e)First episode of epistaxis or bleeding or swelling

Figure 6 shows that the maximum episodes 47.06% (8/17) occurred in the second week followed by 29.41% in the first week.

• (f)Number of bleeding episodes

Fig. 6.

Bar diagram describing the first episode of epistaxis or bleeding after orthognathic surgery. 47.06% occurred in the second week

Mean bleeding episodes were 2.58 ± 0.996.

• (g)Artery involved

The arteries affected were internal maxillary artery (42%), sphenopalatine artery (27.27%), facial artery (15.15%), descending palatine artery (12.12%), internal carotid artery (9.09%) and infraorbital artery (3.03%) as shown in Fig. 7.

• (h)Average time for diagnosis of PAs

Fig. 7.

Bar diagram showing incidence of various arteries involvement in PAs in various orthognathic surgeries

Average time for CT or angiographic diagnosis of pseudoaneurysms within 2 weeks postoperatively as shown in Fig. 8.

• (i)Intervention

Fig. 8.

Bar diagram showing average time for diagnosis of PAs after surgery (CT/angiography)

Embolization was performed either by coils, gelform, etc. in 81.81% cases; ligation of artery was done in 15.15% cases and in one patient surgical resection of the pseudoaneurysm was done.

Discussion

From the review of the literature, it is possible to draw numerous conclusions regarding pseudoaneurysms following the orthognathic surgery. The risk of pseudoaneurysms is much lower in mandibular osteotomies than maxillary osteotomies. In concurrence, most episodes in our review were associated with Lefort I osteotomy (69.7%), followed by sagittal split osteotomy (24.24%) and other surgical procedures (6.06%).

Most reported cases of PAs of external carotid artery (ECA) appear in the superficial temporal and facial artery due to trauma-related injury, as these branches are more vulnerable due to their relatively superficial position [10–12]. In this review, sphenopalatine artery (27.27%) is the most commonly involved branch for PAs. Laskarides et al., on the other hand, pointed descending palatine artery to be most commonly injured branch [12].

The maxillary artery along with its branches is more prone to damage especially at pterygopalatine fossa region when maxillary tuberosity is osteotomized from pterygoid plates or during downfracture procedure [13, 14]. Use of curved osteotomes for pterygomaxillary disjunction is still controversial in this regard. Acebal-Banco et al. described no life-threatening hemorrhages following use of curved osteotome [15]. Precious et al. presented 500 Lefort I procedures without the use of osteotome for disjunction wherein no perioperative bleeding occurred [16]. These hemorrhagic episodes were reported more commonly in correction of syndromic maxillary hypoplasia [17].

In case of mandibular osteotomies, the internal maxillary artery is highly at risk as chances of soft tissue injury are higher when the osteotomy cut is made near sigmoid notch [14]. There are less chances of damage to facial artery even though it is close related with the osteotomy site because of subperiosteal reflection of the tissues that preserves the artery laterally [18]. One case of pseudoaneurysm of internal maxillary artery is also reported in literature following mandibular distraction osteogenesis [19].

Intraoperative blood loss and amount of blood loss in epistaxis or number of episodes is not very well documented in the literature. Only 10 patients out of 33 (30.3%) had values for intraoperative blood loss. We cannot comment on the consensus of intraoperative blood loss with formation of pseudoaneurysm. Moreover, the patient usually gets discharged and may be treated by some other hospital/doctor for emergency bleeding episode or the patient might be having mild episodes, which the patient cannot keep record of. Mean bleeding episodes were 2.58 ± 0.996 as per our review.

Detailed clinical history and physical examination is strongly recommended for diagnosis of pseudoaneurysm. Patient may present with mild to severe nasal/oropharyngeal bleeding or multiple epistaxis episode due to rupture from PAs. Many authors strongly believe that patient will present with bleeding episodes within 2 weeks of surgery [20]. Avealar stated that PAS usually occurs 3 days to 8 months after the osteotomy [21]. Ideally, as stated by Lanigan in 1990, any patient who presents with first episode of epistaxis after second postoperative week or who experiences recurrent significant hemorrhage after orthognathic surgery, should be investigated by angiography to rule out pseudoaneurysm. [14]

The initial absence of pulsation in the mass is a late phenomenon as it does not occur until after liquefaction of the hematoma. A bruit/thrill is common in aneurysm, but the bruit is localized and is generally heard during systole [22]. The lesion is usually soft on palpation, pulsatile and has auditory machinery murmur. This bruit/thrill can be absent in deep lesions especially deep maxillary artery branches, in which palpation becomes difficult although the patient can feel the pulsations [23]. Use of stethoscope should be preferred. Aspiration of the suspicious lesions should be avoided as it can precipitate bleeding.

Differential diagnosis of a pseudoaneurysm include hematoma, abscess, cyst, inflamed lymph node, juvenile nasoangiofibroma and arteriovenous fistula. A detailed history, recent onset, pulsations and epistaxis episodes make pseudoaneurysm as a likely fit.

Radiographs can be of little use in the diagnosis mainly when the wall has become calcified. Angiography and contrast-enhanced CT (CECT) are the preferred radiographic modalities used for diagnosis. CECT was considered as a diagnostic tool but it can be of less value when the size of the lesion is small. Doppler USG can also be used for fast screening when the pathology is superficial. The main advantage, is its capacity to diagnose turbulent fluid with 95% accuracy [24]. An angiogram is better enabled for identifying the vascular injury more quickly and the exact location can be accurately identified allowing precise treatment.

Airway patency in these patients should be the foremost concern as distress from continuous bleeding can further worsen the situation. A combination of conservative and surgical treatment should be initiated to tackle such life-threatening bleeds. Anterior/posterior nasal packing, direct pressure or topical hemostatic agents can be used in a hemodynamically stable patient. Controlling blood pressure and administering intravenous fluids along with blood products are required for an unstable patient.

If PA is diagnosed via angiography, preferred treatment is trans-catheter embolization. In trained hands, Digital subtraction angiography is a safe procedure with a 0.03% mortality and 1.73% morbidity rate which is not outweighed by the risk of uncontrolled bleeding [25, 26]. Surgery can be used to resect the aneurysms or direct ligation of the artery can also performed [6, 8, 9, 27]. Embolization is always advantageous over surgical intervention because it can spare more proximal vessels, and we can selectively embolize the distal bleeding source. Moreover, it has an additional advantage of maintaining the blood supply of the osteotomized segments preventing aseptic necrosis which is particularly important following orthognathic surgery. Substances used in embolization include gelfoam, Gianturco coils, stainless steel coils with gelatin, detachable balloons and various chemicals like PVA, N-butayl cyanoacrylate, etc. [5, 18, 23, 26, 27]. Detachable platinum coils can be easily used to obliterate a large blood vessel without causing adverse effects. These have a rapid hemostatic effect and can be removed readily in case of an incorrect placement [28]. Embolization is contraindicated in patients with allergy to contrast media or severe carotid ateromatosis [7].

More aggressive treatment modalities like trans-antral ligation of IMA become very difficult because of altered anatomy after surgery, postsurgical arterial retraction wherein a tiny bleeding point is often very difficult to spot. Proximal ligation of IMA or ECA may allow collateral filling of distant PA and re-bleeding [26]. Ligation of ECA has been successfully performed by Lanignand 1983 [29]. Surgical excision is the last resort which is not advised these days although Weber et al. 1997 stated the complete excision of pseudoaneurysm along with ligation of both ends of artery as recommended treatment [30].

Due to excessive collateral supply in head and neck region, reoccurrence of the lesion is high as stated by Akai et al. 1994 [31].

Limitation

All the articles in the review are case reports or case series or retrospective studies. Prospective data are required for even minor episodes of epistaxis/bleeding (Table 1).

Table 1.

Literature review of all the data available regarding PAs after orthognathic surgery

Author	Age in years	Sex	Procedure	Intraoperative blood loss	First episode of bleeding/epistaxis	Number of bleeding episodes	Artery	Duration of development PAs (diagnosis)	Intervention
Hemming et al. 1987 [5]	29	Female	Lefort I	–	11th day postoperative	4	Sphenopalatine artery	2 weeks	Gianturco coil
Clark et al. 1987[22]	15	Male	Subcondylar osteotomy	–	–	–	Maxillary artery	9 weeks	3 mm gianturco coil
Solomons and Blumgart 1988 [32]	20	Male	Lefort I	–	–	–	Maxillary artery	1 month	Large gel foam followed by 3 mm gianturco coil
Lanigan et al. 1991 [6]	13 20 29	Female Female Male	Lefort I Lefort I Lefort I	– <500 ml 1000 ml	6 weeks 3 weeks 6 days	– 3 2	Sphenopalatine artery Sphenopalatine artery Maxillary artery	6 weeks 7 weeks 10 days	Direct ligation of artery Direct ligation of artery Embolization
Lustbader DP et al. 1991 [7]	17	Male	Lefort I	–	–	–	Maxillary artery	12 days	Three guide wires
Yin 1994 [8]	24	Male	Sagittal osteotomy	–	–	–	Maxillary artery	During surgery	Multiple ligations
Albernaz 1995 [33]	30	Female	Lefort I	–	–	–	Descending palatine artery	22 days	50 pieces of polyvinyl acrylate
Rogers 1995 [34]	23	Male	High lefort I	–	–	–	Maxillary artery	3 days	Fiber coils
Tiner BD, 1997 [9]	32 35	Male Male	Lefort I Lefort I	– –	2 weeks 8 days	3 1	Descending palatine artery Internal maxillary artery/internal carotid artery Infraorbital artery	1 month 3 months 8 days	Coils surgical resection Ligature clips
Bardley et al. 2002 [35]	17	Male	Lefort I	–	–	–	Maxillary artery	8 months	COIL
Procorpio 2003 [36]	37 24	Male Male	Lefort I Lefort I	– –	– –	– –	Sphenopalatine artery Sphenopalatine artery	63 days 10 days	Platinum coils Platinum coils
Fernandez preito 2005 [26]	26	Female	Lefort I	–	3 weeks	2	Descending palatine artery	1 month	Embolization with microcoils
Manafi 2007 [10]	42	Female	SSRO	–	10 days	3	Maxillary artery	28 days	Embolization with coils
Silva 2007[3]	20	Male	SSRO	1000 ml	–	–	Maxillary artery	6 weeks	Embolization with coils
Elton 2007 [37]	41	Female	Subcondylar osteotomy	–	–	–	Maxillary artery	5 years	Ligation of ECA
Aveolar RL, 2010 [21]	20	Male	Lefort I	500 ml	1 day prior to report	1	Maxillary artery (sphenopalatine artery)	9 weeks	Selective catheterism of sphenopalatine artery and implantation of 7 platinum microcoils
Chepla 2010 [38]	17	Female	Lefort I	–	7 days	–	Internal maxillary artery	14 days	Embolization with microcoils
Madani 2010 [18]	22	Male	SSRO and facial trauma	1000 ml	–	–	Facial artery	4 weeks	Embolization with coils
Krishan, 2011 [1]	33	Male	Lefort I	–	–	–	Internal carotid artery	8 days	Coil embolization
Precious DS, 2012 [16]	32 26 20	Male Male Male	SSRO SSRO SSRO	– 150 ml 1000 ml	2 weeks 1 weeks 1 week	– –	Facial artery Facial artery Facial artery	3 weeks 5 weeks 1 week	Coil embolization Coil embolization Embolization
Kim 2013 [39]	31	Male	Lefort I	–	–	–	Sphenopalatine artery	2 weeks	Embolization
Hacrin Bay 2013 [40]	18	Male	Lefort I	–	–	–	Internal carotid artery	2 days	CA stent reconstruction and PA coil obliteration
HW Jo 2013 [41]	19	Male	SSRO	–	–	–	Facial artery	3 weeks	Embolization with histoacryl
Niazi, 2018 [42]	–	–	Lefort I	–	1 week	–	Descending palatine artery	1 week	Coils
Bykowski et al. 2018 [43]	27	Male	Lefort I	600 ml	2nd week	4	Maxillary artery	36 days	Coil embolization
Laskarides, 2018	22	Female	Lefort I	<150 CC	2nd week	>3	Sphenopalatine artery	21 days	Onyx mediated embolization
Our report, 2019	21	Male	Lefort I	450 ml	8 days 17 days	3 2	Distal branch of maxillary artery Sphenopalatine artery	10 days 17 days	Embolization with gelfoam Embolization with gelfoam

Conclusion

If a patient has recurrent epistaxis or swelling after orthognathic surgery, it is advisable to go for diagnostic imaging like angiography without any delay. In recent times, advanced techniques and expertise are readily available for early diagnosis and management of pseudoaneurysm.

Funding

No sources of support in the form of grants

Compliance with Ethical Standards

Conflict of interest

The authors have no conflict of interest to disclose.