62

ARTERIOVENOUS MALFORMATION OF THE MANDIBLE: A RAREBUT LIFE-THREATENING DISEASE

Baharudin Abdullah, Abdullah Pohchi* & Abdul Rani Samsudin*

Department of Otorhinolaryngology ORL - Head and Neck SurgerySchool of Medical Sciences, *School of Dental Sciences, Universiti Sains Malaysia,

16150 Kubang Kerian, Kelantan, Malaysia

AVM in the mandible is rare. It may present with recurrent episodes of unexplainedgingival haemorrhage, bony swelling, tooth mobility or facial asymmetry. Wereported our experience in managing a case of a 15 year old Malay girl whopresented with a life threatening bleeding from her mandible.

Key words: Arteriovenous Malformation, Mandible

Introduction

AVM in the mandible may present withrecurrent episodes of unexplained gingivalhemorrhage, bony swelling, tooth mobility or facialasymmetry. The most unfortunate presentationhowever follows dental extraction or bone biopsywhen severe, exsanguinating and torrentialhemorrhage may occur. Occasionally, a patient mayonly present with a sentinel molar bleeding whichprecede the hemorrhage and this fact should not beoverlooked (1). Sometimes a bruit can be heard onauscultation over the involved area and a sensationon the region innervated by the mandibular branch

of the trigeminal nerve (V) may be altered.

Case Report

A 15 year old Malay girl from Ipoh, Perakwho experienced persistent severe bleeding from thelower right gum was referred to our hospital. Herproblem started when she was seven years old whos,she experienced bleeding from the lower right firstpremolar tooth. The bleeding was minimal butrecurred which prompted her parents to bring her toa private dental clinic in Ipoh several times. Thebleeding was about 4 to 5 times per year.Unfortunately had the bleeding got worse and never

Submitted-20-02-2004, Accepted-03-12-06

Malaysian Journal of Medical Sciences, Vol. 14, No. 1, January 2007 (62-64)

CASE REPORT

Figure 1 : The orthopanthomogram (OPG) showed a soap bubbleappearance of the right mandible.

63

resolved. She developed persistent bleeding fromthe same region at the age of 15 years and wasbrought to Ipoh General Hospital. The bleedingoccurred spontaneously without any trauma to thatregion. Each bleeding filled up 1 to 2 cupfuls.Applying pressure will only stop the bleedingtemporarily.

On examination there was a diffuse swellingon the outer aspect of the right mandible. Intraorally

there was a bluish lesion in the right retromolartrigone which extended anteriorly to the premolarregion. No bleeding was seen. The throat and theneck were normal. Other systemic examination wasunremarkable. No lesion seen in the nasopharynx,oropharynx and hypopharynx on flexible endoscopy.Her orthopanthomogram (OPG) and CT scanshowed a soap bubble appearance of the right sideof the mandible extending from the right ramus to

ARTERIOVENOUS MALFORMATION OF THE MANDIBLE: A RARE BUT LIFE-THREATENING DISEASE

Figure 1 : The computed tomography (CT) scanof the mandible demonstrated theabnormal right mandible.

Figure 3 : Selective catheterization of the rightlingual artery for embolization.

64

the body of the mandible up to the 1st molar areafigure 1 and figure 2. A diagnosis of AVM ofmandible was made which was subsequentlyconfirmed by magnetic resonance imaging (MRI)and magnetic resonance angiography (MRA). MRIbrain with gadolinium showed no intracranialinvolvement.

Selective embolization of the right lingualartery was performed prior to the right segmentalmandibulectomy (figure 3). A median lower lip splitand right submandibular incision was made. Thecarotid triangle was identified where the externalcarotid artery and its branches were located. A silkstring was placed under the external carotid arteryto secure bleeding in case hemorrhage occursintraoperatively. The periosteal flap of the mandiblewas raised. A tumor mass was seen involving thelateral surface and cortex of the ramus of the rightmandible extending to the second molar tooth region.Feeding vessels were noted along the condyle. Asegmental mandibulectomy was performed byremoving part of the right mandible between theright second premolar and first molar teeth to theright condylar head. The defect was reconstructedby using a 7cm rib graft taken from the right fifthrib. The graft was placed over the defect and fixedto the mandible with reconstructive titanium plateand screws on both sides. Postoperatively the patientwas managed for 1 day in the intensive care unit.She recovered well and was discharged on day 7postoperatively.

Discussion

Arteriovenous malformations (AVM) arisingwithin the mandible is exceedingly rare butpotentially life threatening. The lesion is more oftenlocated in the horizontal portion of the lower jawthan in the condylar process or in thetemporomandibular joint (2).

An OPG may disclose multilocularradiolucent areas with ‘honeycomb’ or ‘soap bubble’appearance or unilocular osteolytic images or simpleradiolucency without peculiar characteristics;sometimes with cortical expansion, dentaldislocation and root resorption of the teeth (2). Lesscommon presentation includes extension of bonyspicules extending at right angles into the lesion(sunburst or sunray appearance) which is highlydiagnostic of AVM (1).

Resection has been advocated both as primarytherapy and as salvage treatment after failure ofconservative measures (3). The indications for

resection suggested are obstruction of visual axis;large lesion with thrombocytopenia; obstruction ofluminal structures; uncontrollable ulceration,hemorrhage or infection; atypical growth suggestingalternative diagnosis; cardiopulmonarydecompensation from arteriovenous shunting and;small lesions that can be excised without cosmeticor functional risk (3).

In this patient resection of the mandible(segmental mandibulectomy) proved to be lifesaving.

Corresponding Author :

Dr. Baharudin Abdullah MBBS(Mal), MMED ORL-HNS (USM)Department of Otorhinolaryngology ORL - Headand Neck Surgery(ORL-HNS),School of Medical Sciences,Universiti Sains Malaysia, Health Campus,16150 Kubang Kerian, Kelantan, MalaysiaTel: + 609-7664110Fax: +609-7653370Email: baharudin@kb.usm.my

Otorhinolaryngology ORL

References

1. Sofferman R.A. and Summers G.W. Bilateralarteriovenous malformation of the mandible.Ann.Otol.Rhinol.Laryngol 1991; 100: 759-767.

2. Maurizi M., Fiumicelli A., Paludetti G. and SimoncelliC. Arteriovenous fistula of the mandible: a review ofthe literature and report of a case. Int J PediatricOtorhinolaryngology 1982; 4: 171-179.

3. Weber T.R., Connors R.H., Tracy T.F. and Bailey P.V.Complex hemangiomas of infants and children. ArchSurg 1990;125:1017-1021.

Baharudin Abdullah, Abdullah Pohchi et. al