Int. J. Oral Maxillofac. Surg. 2001; 30: 397–401 doi:10.1054/ijom.2001.0120, available online at http://www.idealibrary.com on
Clinical paper: Clinical pathology
Conservative treatment of arteriovenous malformations of the mandible
Roberto Brusati, Silvestre Galioto, Federico Biglioli, Mario Goisis Department of Maxillo-Facial Surgery, San Paolo University Hospital, Milan, Italy
R. Brusati, S. Galioto, F. Biglioli, M. Goisis: Conservative treatment of arteriovenous malformations of the mandible. Int. J. Oral Maxillofac. Surg. 2001; 30: 397–401. 2001 International Association of Oral and Maxillofacial Surgeons Abstract. A modified technique is reported on the treatment of arteriovenous malformations in the mandibular body. The technique consists of highly selected embolization and piecemeal removal of the lesion through burred holes made in the cortex. This way the mandible and the teeth may be saved in selected patients.
Intra-osseous mandibular arteriovenous malformations (AVMs) are rare (5% of all vascular malformations) but of great clinical importance, particularly in the case of high-flow AVMs15 (which could lead to lethal haemorrhages)8,9,11,13. Their treatment has been substantially improved by the introduction of sophisticated embolization procedures that considerably reduce the blood flow to the malformation11. However, as in the case of the previous use of simple vascular pedicle ligation20, embolization alone4 is not sufficient to definitively resolve the problem: the presence of residual collateral blood flow may lead to progressive revascularization modifying vascular anatomy, making further embolization much more complex if not impossible5,11,13,15,17,18,22. The first proposed treatment included resection of the involved bony segment and temporary or permanent ligation of the neck vessels. This was followed by prosthetic replacement of the lost bone, reconstruction with free bone grafts6,8,10,14,16,24 or microvascular anastomosed bone 0901-5027/01/050397+05 $35.00/0
grafts7. This treatment often led to massive blood loss. It also led to the loss of teeth in the area involved and required a donor site for the bony reconstruction. In an attempt to obviate this problem, some authors described replacement of the resected bone segment freed of AVM but without the teeth14,23. A considerable step forward in the treatment of these lesions was made by the introduction of the procedure developed by M and A, and others authors12,17,18,19,21. After having ligated the neck vessels, removed a piece of the buccal cortex bone and related teeth, this was followed by curettage of the whole of the bone lesion, thus preserving the continuity of the mandible. Subsequently, with the use of embolization, A et al.2 carried an exclusively intra-oral treatment by extracting the teeth and then cleaning the underlying cavity through the alveolar process and packing the mandible with oxidated cellulose. This procedure, moreover, still resulted in the loss of the teeth involved. It
Key words: vascular malformation; arteriovenous malformation; AVM; mandible; surgery. Accepted for publication 1 April 2001
is the purpose of this paper to report a further improvement of this technique saving the teeth. Operative technique After highly selective embolization performed no more than 24–48 h before the surgical intervention, the lesion is approached using a submandibular route. External carotid, lingual and facial arteries are identified and prepared for clamping in case of neeed during the operation. Taking advantage of the submandibular route, the buccal cortex of the mandible is exposed and a burr of 4 mm in diameter is used to burr a hole through the buccal cortex. Rapid curettage with a small spoon followed by oxidated cellulose packing arrests the bleeding which, depending on the previous embolization, may still be considerable. A second hole is made 1–2 cm to the side of the first and the same manoeuvre is repeated. In this way, a series of holes allows one to treat the whole malformation, while limiting blood loss and respecting both
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Fig. 1. Case 1: Panoramic radiograph of AVM of left mandibular body.
mandibular continuity and tooth integrity. The procedure leaves the mandible looking like a piece of Swiss cheese.
Fig. 2. Case 1: CT scan of AVM.
Case reports Case 1
A 10-year-old boy presented with a purple cutaneous vascular malformation
Fig. 3. Case 1: The angiography demonstrates vascularization of the lesion by inferior alveolar, facial and lingual arteries.
Fig. 5. Case 1: Intraoperative view of the body of the mandible.
in the parotid, cheek and left auricle region. The lesion had been present since birth and had been treated with
Fig. 4. Case 1: The vessels are almost completely stopped after embolization.
Fig. 6. Case 1: Radiographic control of the mandible 12 months after surgery, showing complete recovery of the bony structure.
Mandible AVMs
Fig. 7. Case 1: CT control of the mandible 12 months after surgery.
cortisone at various times but did not regress. For approximately one year his relatives had noted facial asymmetry due to
increase in size of the left mandibular body, without pain or tooth mobility. Intra-oral examination revealed a swelling of the entire buccal cortex of the
Fig. 8. Case 2: Large AVM of the face and right mandible.
Fig. 9. Case 2: The arteriography shows the vascular supply from inferior alveolar and facial arteries.
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mandibular body but without any appreciable pulsations. A panoramic radiograph showed a sharp-edged radiolucent lesion consisting of three main roundish images, which extended from the angle of the mandible to the homolateral lateral incisor and was in contact with the roots of all of the teeth involved (Fig. 1). CT with contrast medium revealed a broad osteolytic area within the thinned mandibular cortical walls which was characterized by irregular, intense enhancement (Fig. 2). In order to confirm the diagnostic suspicion of AVM, a thin burr (1 mm diameter) was used to make a hole in the cortex corresponding to the lesion from which arterial blood flowed. This was done using local anaesthesia. The hole was immediately filled with bone wax. Angiography revealed a lesion suspicius to a high-flow AVM fed by the left inferior alveolar artery and, to a lesser extent, by the homolateral facial and lingual arteries. Venous drainage occurred through markedly dilated veins leading to the external jugular vein. The lesion also involved the skin of the region of the mandibular angle, the auricle and part of the temporal bone, with minor multiple vessels arising from the middle meningeal artery, the posterior auricular artery and small branches arising from the internal maxillary artery (Fig. 3). The posterior auricular, middle meningeal, lingual, facial and
Fig. 10. Case 2: The vessels are interrupted by embolization.
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Fig. 11. Case 2: Intraoperatory view of the mandible treated by the described technique.
Fig. 13. Case 2: OPT immediately after surgery.
Fig. 12. Case 2: Intraoperatory view of the mandible treated by the described technique. Note the holes of the cortical bone filled by oxidated cellulose.
Fig. 14. Case 2: OPT 12 months after surgery, showing a good bone healing and spontaneous reintrusion of molars.
inferior alveolar arteries were embolized by means of highly selective catheterization and polyvinyl alcohol (PVA) particles of 150–500 microns in diameter. The embolization considerably reduced flow in the mandibular body, but was less successful in the remaining components of the lesion (Fig. 4). Twenty-four hours later, the patient underwent the procedure described, without significant blood loss (Fig. 5). After 24 months of clinical, radiological and angiographic follow-up, there was perfect ossification of the mandibular medullary bone with no signs of relapse. No treatment-induced damage to the teeth or impair alveolar nerve was observed (Figs 6 & 7). Case 2
A 15-year-old male referred to us for repeated bleedings in the right mandibular molar region. The oral cavity showed purple colour of the right retro molar mucosa, with pulsation clearly appreciable by finger-touch. A clearly pulsating
3 by 5 cm mass was also present in the submandibular region (Fig. 8). The ipsilateral first molar was mobile. CT with contrast medium revealed a round-shaped sharp-edged osteolytic area at the level of the right mandibular angle and body. The lesion was characterized by irregular intense enhancement. Angiography revealed a high-flow AVM involving the right submandibular gland, fed by branches of the homolateral inferior alveolar and facial arteries. Venous drainage took place through the facial and jugular veins. The internal maxillary, transverse facial, facial and lingual arteries of the right side were embolized by means of highly selective catheterization and PVA particles of 600–1000 microns, which led to a nearly complete blockage of the blood flow in the lesion (Figs 9 & 10). Twentyfour hours later, through a submandibular approach, the patient underwent the procedure described, after isolation and removal of the submandibular gland in order to obtain a radical treatment of
the region involved by the lesion. No significant blood loss occurred during surgery (Figs 11 & 12). Three months after surgery, the right inferior first molar was vital and firm again. The 24 month clinical and radiological follow-up revealed satisfactory condensation of the mandible with no signs of recurrence or functional deficits of the involving teeth and the inferior alveolar nerve (Figs 13 & 14). Discussion As indicated by M and A in 196817,18, the fundamental principle in the treatment of AVMs is complete removal. When this is not achieved, recurrence is almost guaranteed and this may lead to gradual worsening due to the regrowth of the residual pathological tissue15. Embolization should, therefore, only be considered as a complementary treatment because it does not lead to elimination of the pathological tissue but simply interrupts the main blood flow11,13,15. Any delay from
Mandible AVMs embolization to surgery exceeding 48 h may lead to greater risk of serious bleeding due to increased blood flow through collateral vessels feeding the lesion13,15. The initially proposed treatment including ostectomy of the mandible usually leads to considerable morphofunctional deficits. Reconstruction of the mandible by means of a bone graft certainly represents a step forward in the improvement of the procedure3,6,10,16,24. In case this was done at the same time of resection, however, some failures occurred because of graft infection due to the possible communications with the oral cavity. This risk can be reduced by delaying the reconstruction for some months8. Microsurgical reconstructions may give rise to more reliable results, but would also increase operating time and lead to more donor site morbidity7. Although the treatment proposed by J et al.14, which involves the replacement of the resected portion of the mandible after having removed its pathological content, avoids the need for bone harvesting, it still leads to tooth loss and the risk of bone graft infections. The treatment suggested by A et al.2 has the advantage that only the teeth close to the lesion need to be extracted, and the pathological tissue is removed through the sockets in the alveolar process. This is best performed in the presence of intact lingual and buccal cortex in order to ensure adequate curettage and a tight packing of the cavity. Revascularization of any small residues of pathological tissue is then avoided. The technique described here offers the additional advantage of preserving the teeth. A prerequisite for success of curettage and tight packing is the presence of lingual and buccal cortex (even if lingual periosteum can be a sufficient barrier for curettage and packing). In our limited experience, the surgical procedure has only been applied through a submandibular approach. In one case because of the association of extension of AVM into the submandibular space, in the other one because the incomplete
embolization did not offer sufficient guarantees against bleeding. Yet, a complete intra-oral approach would seem possible in selected patients.
14.
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Address: Dr Pr. Roberto Brusati Cattedra e Divisione di Chirurgia Maxillo-Facciale Ospedale S. Paolo Via A. di Rudinı` 8 20142 Milano Italy Tel: +39 02 81844707 Fax: +39 02 81844704