CASO CLÍNICO

 

Arteriovenous fistula of the lower lip. Case report

Fístula arteriovenosa del labio inferior. Presentación de un caso

 

 

M. Estrada Sarmiento¹, L.I. Virreyes Espinosa², S. Gonzáles Pardo³

1 Profesor auxiliar. Especialista de II grado Cirugía Maxilo Facial. Presidente de la Filiar Granma de Cirugía Maxilo Facial. 
Asesor de la Universidad Virtual de Salud.
2 Licenciada en enfermería Jefa del área quirúrgica.
3 Especialista de I grado en anestesia y reanimación. Vice director quirúrgico. Granma, Cuba

Correspondence

 

 

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ABSTRACT

The object of this work is to present our experience in the surgical management of an arteriovenous fistula in the lower lip of a fifteen year-old patient as a result of a bite. The patient was diagnosed initially as having a trauma-induced hemangioma, which was treated with steroid and surgical exeresis. Four years later, a larger tumor appeared. The external carotid artery was dissected and an arteriography of the carotid was carried out that gave the diagnosis of arteriovenous fistula. Ligation was carried out of the facial artery and of the venous vessels for isolation from the circulation. Four injections were given with 4cc of betamethasone into the mass. Later 4 additional injections were given with intervals of 3 weeks. Four months after the ligation of the vessels, the fibrous mass was excised. Five years have now passed since the last intervention and there has been no recurrence. The importance of the angiography should be highlighted for diagnosing and treating this very rare pathological entity.

Key words: Arteriovenous fistula; Angioma; Vascular malformation.

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RESUMEN

El objetivo del trabajo es exponer nuestra experiencia en el tratamiento quirúrgico de una fístula arteriovenosa del labio inferior de una paciente de 15 años de edad, producida por un mordisco. La paciente fue diagnosticada en su inicio como un hemangioma traumático, el cual fue tratado con esteroides y exéresis quirúrgica. A los 4 años apareció con una tumoración más voluminosa, se realizó disección de la carótida externa y arteriografía carotídea la cual diagnóstico fístula arteriovenosa, se realizó ligadura de la facial y de los vasos venosos, para aislarla de la circulación. Se inyectaron 4 cc de betametazona en la tumoración, posteriormente se inyectaron 4 inyecciones adicionales con intervalos e 3 semanas, a los tres meses de la ligadura de los vasos sé realizo la exéresis de la fibrosis de la tumoración. En estos momentos la paciente tiene 5 años desde la última intervención, no presentando recidiva. Se destaca la importancia del estudio angiográfico para el diagnóstico y tratamiento de esta entidad patológica, que es poco frecuente.

Palabras clave: Fístula arteriovenosa; Angioma; Malformación vascular.

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Introduction

An arteriovenous fistula is an abnormal communication between an artery and a vein. It can be congenital or acquired.¹ Those that are congenital are extremely rare and 50% occur in the area of the head and neck. The acquired form is produced by trauma leading to broken blood vessels. On healing, an artery communicates with a vein and the normal blood flow is inverted. As a result of an increase in blood pressure in the affected area, the vessels dilate appearing to be a cavernous hemangioma or an aneurisma.^1,3 This lesion has been a challenge with regard to treatment, as its surgical removal is a technical problem, as is the anatomic restoration and function of the area affected. ⁴ When operated it has to be extensively resected or the possibilities of recurrence are very high, as collateral circulation is created that is very ample, and this worsens the symptoms.⁴

Trauma-induced arteriovenous fistulas of the lower lip are very rare. In the medical literature reviewed, we have only found one case published in Cuba by Felipe Rodríguez that is similar to ours.The rarity of this condition motivated us to review the literature and produce this work based on the experience acquired over nine years with a patient with this pathological entity.

Clinical characteristics

The local symptoms are:

• Murmur.
• Thrill.
• Phlebectasia.
• Hyperthemia.

Diagnosis. We should consider an arteriovenous fistula whenever a murmur and thrill appear in a region previously affected by a penetrating wound.

Differential diagnosis. The arteriovenous fistula should be differentiated from:

• Venous malformations: These are soft masses, that are pulsatile and that can be compressed. These lesions grow proportionally with the patient and increase on compressing the jugular vein or with Valsalva´s maneuver.

• Lymphatic malformations: These are congenital and many are observed at birth.

• Hemangiomas: These are vascular tumors with a growth phase that is marked by endothelial and cellular proliferation and an involution phase.

 

Case Report

Female patient, RGP, medical record Nº 68 14 58. The reason given for admittance was an increase in volume of her lower lip.

The condition started at the age of 15 after her lower lip was bitten, which resulted in intense hemorrhaging. As a result of the trauma, the middle and right side of the lip swelled progressively. The area was nontender, purpleblue in color, pulsatile and with a diameter of 3 cm. It was classified as a traumainduced hemangioma and it was treated with betamethasone injections. After 6 doses of betamethasone, the mass was removed using a buccal approach. The histopathologic diagnosis was compatible with a trauma-induced hemangioma.

Two years after being treated the patient presented again, as she had a larger increase in the volume of the lower lip. The area was pulsatile and there was a murmur and thrill (Fig. 1).

The intra-buccal examination revealed a raised vascular pattern in the mucosa of the lower lip (Fig. 2) An ultrasound was requested of the region and dissection was carried out of the right carotid artery for an arteriography, which showed an aneurismatic bulge by the lower labial and chin region (Fig. 3 and 4). The external carotid artery was ligated between the upper thyroid and lingual arteries (Fig. 5). The venous vessels of the aneurysm were ligated by means of a submental incision in order to isolate the cavity from the circulation.

At the end of the surgery, 4cc of betamethasone was injected into the mass and elastic sparadrap was applied. Four additional injections of triamcinolone were given with three week intervals. The progressive reduction together with the fibrosis of the mass was observed, and no murmur was detected.

Three months after the ligation of the vessels, the lip mass was excised. The final aesthetic result was acceptable (Fig. 6A and B). Five years have elapsed since the surgery and there has been no recurrence of the lesion.

 

Discussion

Arteriovenous lesions can have a traumatic or a congenital origin. Traumatic fistulas make up 7.8% of all vascular lesions.5 In various studies published a traumatic origin was found in approximately 90% of cases.^5,6 The prognosis depends fundamentally on the size of the fistula and its location. The symptoms can be local or general.5 Angiographic and ultrasound tests will provide the diagnosis and the exact location of the fistula.^7-11

We revised the national literature but found only one case, presented by Felipe Rodríguez¹ that was similar to ours. The case had arisen as a result of a fall and it had a 15-year course.

The failure of the first operation was due to a lack of knowledge as to the circulatory aspect of the lesion. The carotid arteriography permitted establishing a diagnosis of arteriovenous fistula. It permitted knowing the area with the shunt and it provided information on the anatomic shape of the lesion, which was very important information for treating it surgically.

Our case had a clinical course of 7 years. Currently 9 years have passed since the last intervention. There has been no recurrence and the aesthetic result is acceptable.

Arteriovenous fistulas of the lower lip are very infrequent and their surgical treatment is a challenge for surgeons.

 

 

Correspondence:
Manuel Estrada Sarmiento
Máximo Gómez 6 / maceo y Canducha Figueredo.
Bayamo CP: 85100 Granma, Cuba
Email: mesarmiento.grm@infomed.sld.cu

Recibido: 01.06.05
Aceptado: 06.10.06

 

References

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8. Ziyeh S, Schumacher M, Strecker R. Head and neck vascular malformations: time-resolved MR projection angiography. Neuroradiology 2003;45:681-6.        [ Links ]

9. Thomas JA, Ware TM, Counselman FL, Surgical treatment of Traumatic fistula of the lower lip. J Emerg Med 2002;22:257-61.        [ Links ]

10. Harada J, Mino Y, Hasegawa S, Kubo M, Kuwayama N, Endo S. Traumatic arteriovenous fístula of the lower lip caused by an injury due to penetration by pieces of glass: case report. Oral Maxillofac Surg Clin North Am 2002;30:205-8.        [ Links ]

11. Adame N Jr, Bayless P. arteriovenous fistula in the lip as a result of a bite. J Emerg Med 1998;16:575-8.        [ Links ]