|Year : 2010 | Volume
| Issue : 1 | Page : 45-49
Arteriovenous malformation of mandible: Extracorporeal curettage with immediate replantation technique
Virendra Singh, Pramod Kumar Bhardwaj
Department Oral & Maxillofacial Surgery, Government Dental College, PGIMS, Rohtak, Haryana, India
|Date of Web Publication||9-Sep-2010|
Pramod Kumar Bhardwaj
Department Oral & Maxillofacial Surgery, Government Dental College, PGIMS, Rohtak, Haryana
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Arteriovenous malformations of jaw are extremely rare conditions that can result in disastrous complications, if handled carelessly. Although various treatment modalities have been advocated in the literature, there seems to be no complete consensus on a suitable treatment in these cases. This report highlights the importance of correct diagnosis and early treatment in management of vascular malformations. Extracorporeal curettage followed by immediate replantation yielded good results in our case and this technique can emerge as a valid alternative, especially in developing countries.
Keywords: Arteriovenous malformations, extracorporeal curettage, jaw
|How to cite this article:|
Singh V, Bhardwaj PK. Arteriovenous malformation of mandible: Extracorporeal curettage with immediate replantation technique. Natl J Maxillofac Surg 2010;1:45-9
|How to cite this URL:|
Singh V, Bhardwaj PK. Arteriovenous malformation of mandible: Extracorporeal curettage with immediate replantation technique. Natl J Maxillofac Surg [serial online] 2010 [cited 2022 May 19];1:45-9. Available from: https://www.njms.in/text.asp?2010/1/1/45/69168
| Introduction|| |
Arteriovenous malformations (AVMs) of the mandible are rare and challenging entities. The lesion can present with a variety of clinical findings depending on the severity of malformation. Interosseous AVM often remains undiagnosed until a dramatic bleeding incident occurs, which is usually a result of dental manipulation. AVMs have a high propensity to bleed, which may be life threatening. 
Various treatment modalities have been described for treatment of high flow AVM, most of them should be adjunct to surgery. Resection of the mandibular fragment containing lesion has been considered essential for complete healing.  Curettage of the resected fragment with immediate replantation reduces the morbidity associated with the procedure and the difficulty of reconstruction. This seems to be a good approach.
Following the above logic, here we present a case where AVM of mandible was treated with extracorporeal curettage of lesion and immediate replantation of segment.
| Case Report|| |
A 16-year-old female patient reported to emergency department with a massive bleeding episode after extraction of tooth, which could only be controlled after biting on gauze piece. Bleeding stopped only after applying sustained local pressure over the extraction site. History revealed that left mandibular second premolar was carious and associated with persistent pain and swelling in the region for the past 2 years. When swelling did not subside even after taking antibiotics, local dentist extracted the tooth without taking any radiographs, resulting in massive uncontrolled bleeding. On detailed examination after 12 hours, a soft, pulsatile, tender swelling with audible bruit was noted in left submental region [Figure 1]. The overlying skin was normal in color and there was no paresthesia of the involved region. Intraoral examination revealed extraction socket of 35; mobile 34, 36. Marginal gingival bleeding was also noted i.r.t 33, 34, 36, 37 with buccal expansion of mandible. Rest of the physical examination was unremarkable and other blood investigations were normal. At this stage, the patient refused treatment and returned after few months; meanwhile, swelling had increased to the present size and the patient had suffered from two bleeding episodes.
Colored ultrasonography of soft submental swelling revealed high flow lesion with multiple feeders. Orthopantmograph (OPG) showed extrusion of 33, 34 with displacement and tilting of adjacent teeth. The ill-defined moth eaten type radiolucency involved left mandibular parasymphysial body region; also inferior alveolar canal was dilated and tortourous with enlarged mental foramen [Figure 2]. Axial, coronal sections of computed tomography (CT) scan revealed multiseptae central bony destructive lesion with expansion of bicortical plates without perforation [Figure 3]a, b. Needle aspiration from some distance away from the lesion resulted in syringe full of bright red blood. The patient was diagnosed to be a case of AVM mandible.
|Figure 2 :Preoperative OPG showing moth eaten irregular radiolucency in left mandibular parasymphysis body region with enlarged and torturous mandibular canal; adjacent teeth are displaced|
Click here to view
|Figure 3 :a, b Axial CT and Coronal CT section demonstrating widespread destruction and thinning of cortical plates.|
Click here to view
The patient was taken to the operating room and elective tracheostomy was done to maintain airway postoperatively in case of massive swelling. Hypotensive general anesthesia was maintained producing systolic pressure of approximately 70 mm Hg. A wide exposure was attained with large submandibular incision extending to submental region. Dissection was done to identify common carotid artery. After achieving proximal and distal control, the external carotid artery was temporarily ligated above superior thyroid artery. Engorged vessels supplying to AVM were identified and ligated. Osteotomy cuts were given in buccal cortical plates away from the lesion, and using bone spreader, inferior alveolar artery was identified and ligated. Three units of blood were transfused during resection of the segment. Using extracorporeal technique, teeth were extracted in the involved segment, followed by curettage and hollowing of mandible with curretes, rotating handpiece and bur [Figure 4] and [Figure 5]. No graft was placed inside the cavity. Stabilization of reimplanted mandibular piece was done with three 2.5 mm miniplates [Figure 6]. Intermaxillary fixation was done on the contralateral side to achieve occlusion and strength. Postoperative course was uneventful. During follow up of 2 years, ultrasound revealed no new vascular lesion in the region while excellent facial form and function was maintained [Figure 7] and [Figure 8].
|Figure 5 :Extracorporeal technique of extraoral removal of teeth and curettage of lesion|
Click here to view
|Figure 6 :Reimplantation of mandibular segment and stabilization with miniplates|
Click here to view
|Figure 7 :Postoperative OPG showing good alignment of mandibular lower border and fixation with plates at symphysis and angle|
Click here to view
|Figure 8 :Postoperative radiograph showing maintenance of normal anatomical contour|
Click here to view
| Discussion|| |
International Society for the Study of Vascular Anomalies has classified them as hemangioma (with endothelial proliferation) and vascular malformation (VM) (with normal endothelial tumor) relying on 1982 biologic classification of Mulliken and Glovacki  based on endothelial characteristics. The VMs can be further categorized as low flow lesions (capillary, venous, lymphatic malformations) and high flow lesions (AVMs, arteriovenous fistulae) according to blood flow characteristics.  AVMs are most common high flow lesions.
Intraosseous VMs of the maxillofacial region sometimes give rise to dental emergencies and may cause disfigurement, morbidity and even death. , The proximity of the teeth can prove disastrous, like in our case where extraction of tooth resulted in massive hemorrhage. A review of fatal cases by Lamberg and others  shows that in most instances, exsanguination is the result of dental extractions, the dentist being unaware of existence of the AVM.
Vascular lesions of the jaws have an overall 2:1 female:male occurrence, with peak incidence in the second decade. , In this female patient of 16 years of age, persistent pain and mandibular swelling from the past 2 years were main concerns initially. Although more than 50% of vascular lesions occur in the head and neck region, only a small percentage of these occur in jaws. ,, They are twice as common in the mandible as in the maxilla.  Mandibular VM usually appears during adolescence, with extremes at 3 months and 74 years of age.  VMs are caused by a disturbance in the late stages of angiogenesis (truncal stage) and result in the persistence of AV anastomosis present during embryonic life. VMs, which usually present as developmental anomalies from birth, develop in proportion to physical growth. The increase in size of these VMs is asymptomatic and imperceptible at an early age and is promoted by local hemodynamic factors. The blood shunted to the malformation causes the lesion to grow, which in turn causes increased shunting of the blood, hence leading to a vicious cycle. This overgrowth can result from hormonal imbalances, vasomotor disturbances, infections or trauma. 
As has been seen in this lesion, it can present with a variety of clinical manifestations depending on the severity of the malformation; however, occasionally asymptomatic cases have also been reported in patients with AV malformation. , Some of the signs and symptoms reported are soft tissue swelling,  paresthesia, , pain of variable intensity, ,,,, teeth mobility and migration, ,,,, discoloration of overlying skin and intraoral mucosal surfaces, ,,,, facial asymmetry, , local pulsation, ,, noticeable bruit, ,,,, erythematous gingival and bleeding around the teeth , and bone resorption with palpable thrill as well as resorption of the roots in the affected area with no evident tooth-related cause or periapical pathoses. ,, Systemic findings like blurred vision, epistaxsis, paresthesia and cardiac abnormalities (murmur, hypertrophy and failure) have been reported. ,,
The radiographic appearance is quite variable and therefore unreliable as a sole basis of diagnosis. Gelfand  has summarized three typical radiographic appearances:
- A sunray appearance created by trabecular bone between the vessels and osetolytic lesion.
- A soap bubble or honeycomb appearance with occasional punched out areas.
- An appearance described as ill-defined radiolucency.
Other radiographic findings may include cortical expansion as well as unilocular or multilocular cystic areas. Phleboliths, root resorption and lack of lamina dura have also been described.  According to Stafne,  radiographic appearance can resemble any destructive lesion of bone.
Numerous treatments in varying combinations and various degrees of success have been employed, including ligation, , embolization, ,, radical resection,  use of sclerosing solutions,  curettage and packing,  radiation,  bone wax packing in cavities followed by curettage, in two cases observation , and even cryosurgery. ,
Embolization followed by surgical treatment is still the modern conventional approach.  Embolization reduces blood flow, allowing excision to be performed subsequently within 48 hours-2 weeks. , Embolization is also not without risk as embolic complications, allergic reactions, avascular necrosis of bone,  delayed root development,  defective mandibular growth have been reported. Resection of mandible can result in a variety of disabilities including impairment of speech articulation, salivary control, difficulty in swallowing, trismus and deviation of mandible toward the surgical side during functional movement.  This led to the concept of immediate reconstruction which was introduced by Weaver et al. who used the patient's own prefrozen mandibular bone. , Motamedi et al, used this technique in AVM without freezing or autoclaving. The hollowed cortical shell acts as autogenous inductive tray. Greene et al, advocate filling of particulate marrow in the cavity. As digital subtraction angiography and embolization treatment modality are not available in our institute, we had to rely on traditional method of careful ligation of feeder vessel to prevent excessive blood loss, and intraoperative blood infusion was done to prevent any complications.
This young female patient was followed up for almost 2 years and a good esthetic, facial symmetry and functional status was maintained. We have arrived at similar conclusion that this technique is safe, convenient and effective alternative to treat such vascular malformations and restore near exact form, function and symmetry without obviating the need for space maintainers, bone harvesting, future major reconstructive operations and keeping cost factor in check. Resection of involved portion followed by curettage and reimplantation can emerge as a valid alternative to secondary reconstructive surgery.
| References|| |
|1.||Lam SM, Dahiya R, William EF 3rd. Management of an arteriovenous malformation. Arch Facid Plast Surg 2003;5:334-7. |
|2.||Flandroy P, Pruvo JP. Treatment of mandibular arteriovenous malformation of the mandible via resection and immediate replantation of the segment: A case report. J Oral Maxillofac Surg 1997;55:79. |
|3.||Mulliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: A classification based on endothelial characteristics. Plast Reconstr Surg 1982;69:412-22. [PUBMED] |
|4.||Burrows PE, Mulliken JB, Fellows KE, Strand RD. Childhood hemangiomas and vascular malformations: Angiographic differentiation. AJR Am J Roentgenol 1983;141:483-8. [PUBMED] [FULLTEXT] |
|5.||Lamberg MA, Tasanen A, Jaaskelainen J. Fatality from central hemangioma of the mandible. J Oral Surg 1979;37:578-84. |
|6.||Engel JD, Supancic JS, Davis LF. Arteriovenous malformation of the mandible: Life-threatening complications during tooth extraction. J Am Dent Assoc 1995;126:237-42. [PUBMED] [FULLTEXT] |
|7.||Willis RA. Pathology of Tumors. St Louis, MO: CV Mosby; 1948. |
|8.||Stanley E. A treatise on diseases of bone. Philadelphia, PA: Lea and Blanchard Co; 1849. |
|9.||Shira RB, Guernsey LH. Central cavernous hemangioma of the mandible: Report of a case. J Oral Surg 1965;23:636-42. [PUBMED] |
|10.||Watson WL, Mc Carthy WD. Blood and lymph vessel tumors. Surg Gynecol Obstet 1940;71:569. |
|11.||Johnson LM, Cook H, Friedlander A. Central arteriovenous malformations of the maxillofacial skeleton: A case report. J Oral Maxillofac Surg 1991;49:759-63. [PUBMED] [FULLTEXT] |
|12.||Hall EH. What you don't see can hurt you. N Y State Dent J 1993;59:45-7. [PUBMED] |
|13.||Kelly DE, Terry BC, Small EW. Arteriovenous malformation of the mandible: Report of case. J Oral Surg 1977;35:387-93. [PUBMED] |
|14.||Babin RW, Osbon DB, Khangure MS. Arteriovenous malformations of the mandible. Otolaryngol Head Neck Surg 1983;91:366-71. [PUBMED] |
|15.||Hayward JR. Central cavernous hemangioma of the mandible: Report of four cases. J Oral Surg 1981;39:526-32. [PUBMED] |
|16.||Sadowsky D, Rosenberg RD, Kaufman J, Levine BC, Friedman JM. Central hemangioma of the mandible: Literature review, case report, and discussion. Oral Surg Oral Med Oral Pathol 1981;52:471-7. [PUBMED] |
|17.||Halazonitis JA, Kountouris J, Halazonitis NA. Arteriovenous aneurysm of the mandible. Report of a case. Oral Surg Oral Med Oral Pathol 1982;53:454-7. [PUBMED] |
|18.||Greene MW, Nishioka GJ, Triplett RG, Holt GR. Arteriovenous malformation of the mandible: Survey of previously unreported cases and case report. J Oral Maxillofac Surg 1989;47:1322-7. [PUBMED] |
|19.||Anderson JH, Grisius RJ, McKean TW. Arteriovenous malformation of the mandible. Oral Surg Oral Med Oral Pathol 1981;52:118-25. [PUBMED] |
|20.||Maurizi M, Fiumicelli A, Paludetti G, Simoncelli C. Arteriovenous fistula of the mandible: A review of the literature and report of a case. Int J Pediatr Otorhinolaryngol 1982;4:171-9. [PUBMED] |
|21.||Holt GR, Tinsley PP Jr, Aufdemorte TB, Steed DL, Dittman WI. Arteriovenous malformation of the mandible. Otolaryngol Head Neck Surg 1983;91:573-8. [PUBMED] |
|22.||Babin RW, Osbon DB, Khangure MS. Arteriovenous malformations of the mandible. Otolaryngol Head Neck Surg 1983;91:366-71. [PUBMED] |
|23.||Davies RM, Marsden CW, Turner EP. Oral manifestations of an arteriovenous anastomosis. Oral Surg Oral Med Oral Pathol 1977;44:2-6. [PUBMED] |
|24.||Gallagher DM, Hilley D, Epker BN. Surgical treatment of an arteriovenous malformation of the mandible in a child. A case report. J Maxillofac Surg 1983;11:279-83. [PUBMED] |
|25.||Coleman CC Jr. Diagnosis and treatment of congenital arteriovenous fistulas of the head and neck. Am J Surg 1973;126:557-65. [PUBMED] |
|26.||Malan E, Azzolini A. Congenial arteriovenous malformations of the face and scalp. J Cardiovasc Surg (Torino) 1968;9:109-40. [PUBMED] |
|27.||Coleman CC Jr, Hoopes JE. Congenital arteriovenous anomalies of the head and neck. Plast Reconstr Surg 1971;47:354-64. [PUBMED] |
|28.||Gelfand G, Dixon RA Jr, Gans BJ. Central cavernous hemangioma of the mandible. J Oral Surg 1975;33:448-53. [PUBMED] |
|29.||Stafne EC. Oral roentgenographic diagnosis. 3rd ed. Philadeplia: W.B Saunders Company; 1969. p. 198. |
|30.||Moose SM. Sinusoidal aneurysm of the mandible: Report of a case. J Oral Surg (Chic) 1957;15:245-7. [PUBMED] |
|31.||Selfe RW, Sherman M, Miller TF. Arteriovenous malformation of the mandible. Otolaryngology 1978;86:659-66. |
|32.||La Dow CS Jr, Tatoian JA Jr, Lin SR. Treatment of central hemangioma of the maxilla by embolization: Report of case. J Oral Surg 1976;34:622-6. [PUBMED] |
|33.||Kiyosue H, Mori H, Hori Y, Okahara M, Kawano K, Mizuki H. Treatment of mandibular arteriovenous malformation by transvenous embolization: A case report. Head Neck 1999;21:574-7. [PUBMED] [FULLTEXT] |
|34.||Gamez-Araujo JJ, Toth BB, Luna MA. Central hemangioma of the mandible and maxilla: Review of a vascular lesion. Oral Surg Oral Med Oral Pathol 1974;37:230-8. [PUBMED] |
|35.||Lund BA, Dahlin DC. Hemangiomas of the mandible and maxilla. J Oral Surg Anesth Hosp Dent Serv 1964;22:234-42. [PUBMED] |
|36.||Rothman G, Sherman PM, Stoopack JC. Arteriovenous malformation: A five-year follow-up. J Oral Surg 1977;35:936-8. [PUBMED] |
|37.||Maw AR. Some features of arteriovenous malformations in the head and neck. Laryngoscope 1972;82:785-95. [PUBMED] [FULLTEXT] |
|38.||Mohammadi H, Said-al-Naief NA, Heffez LB. Arteriovenous malformation of the mandible: Report of a case with a note on the differential diagnosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:286-9. [PUBMED] [FULLTEXT] |
|39.||Schneider C, Wagner A, Hollmann K. Treatment of intraosseous high flow arteriovenous malformation of the mandible by temporary segmental ostectomy for extracorporal tumor resection: A case report. J Craniomaxillofac Surg 1996;24:271-5. [PUBMED] |
|40.||Yeoman CM. Management of hemangioma involving facial, mandibular and pharyngeal structures. Br J Oral Maxillofac Surg 1987;25:195-203. [PUBMED] |
|41.||Beumer J, Curtis T, Firtell D. Maxillofacial rehabilitation. St Louis: The C.V Mosby Company; 1979. |
|42.||Weaver AW, Smith DB. Frozen autogenous mandibular stent-graft for immediate reconstruction in oral cancer surgery. Am J Surg 1973;126:505-6. [PUBMED] |
|43.||Sakoda S, Shiba R, Irino S. Immediate reconstruction of the mandible in a patient with ossifying fibroma by replantation of the resected segment after freezing. J Oral Maxillofac Surg 1992;50:521-4. [PUBMED] |
|44.||Behnia H, Motamedi MH. Treatment of central arteriovenous malformation of the mandible via resection and immediate replantation of the segment: A case report. J Oral Maxillofac Surg 1997;55:79-84. [PUBMED] [FULLTEXT] |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
|This article has been cited by|
||Uncontrolled bleeding during tooth extraction from an undiagnosed arteriovenous malformation
| ||Kumar Nilesh,Swenil Shah,Amol Gautam,Sagar Thorat |
| ||BMJ Case Reports. 2021; 14(8): e236983 |
|[Pubmed] | [DOI]|
||Arteriovenous malformation of the jaws: a black hole for the GDP – a case report
| ||Kunal Shah,Badrinarayan Srinivasan,Madanagopalan Ethunandan,Clive Pratt |
| ||Dental Update. 2017; 44(5): 444 |
|[Pubmed] | [DOI]|
||An unusual case of intraosseous vascular malformation of the maxilla mimicking fibrous dysplasia: a case report and literature review on imaging features of intraosseous vascular anomalies of the jaw
| ||J-E Kim,W-J Yi,M-S Heo,S-S Lee,S-C Choi,K-H Huh |
| ||Dentomaxillofacial Radiology. 2014; 43(7): 20130400 |
|[Pubmed] | [DOI]|