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Revista Española de Cirugía Oral y Maxilofacial

On-line version ISSN 2173-9161Print version ISSN 1130-0558

Rev Esp Cirug Oral y Maxilofac vol.28 n.3 Barcelona May./Jun. 2006

 

PÁGINA DEL RESIDENTE

 

What should the diagnosis be?

¿Cuál es su diagnóstico?

 

 

Female patient, 52 years old, with no previous history of interest, was referred to our department by her health center dentist as she had a tumor-like mass in the right mandible. It was not painful, nor was it causing any changes in sensitivity. The panoramic radiograph showed a radiolucent image within the jaw that stretched from tooth 4.1 to 4.5. Her physical examination showed a bulging of the jaw, with no dental movement, nor pain to dental percussion, nor any apparent disturbance to the oral mucosa.

The CAT scan showed a lesion in the right anterior part of the mandible, in the basal portion, which measured 3 x 1.7 centimeters. Internal margins could be seen with an apparent geographic pattern and ill-defined borders that was casing a thinning of the adjacent cortex. It appeared to have internal trabeculae. The anterior border of the mandibular lesion had "sun ray" periosteal reaction. There was no evidence of the mass in the soft tissue, nor did any roots show disturbance. The differential diagnosis did not include cystic bone lesions given the periosteal reaction. There was no suspicion of malignancy, as soft tissue was not affected.


Intraosseous hemangioma of the mandible. An intraoral approach

Hemangioma intramandibular. Abordaje intraoral

 

 

R. Luaces Rey1, A. García-Rozado González2, J.L. López-Cedrún Cembranos3, J. Ferreras Granado2, E Charro Huerga1

1 Médico Residente
2 Médico Adjunto
3 Jefe de Servicio
Servicio de Cirugía Oral y Maxilofacial.
Centro Hospitalario Universitario Juan Canalejo, A Coruña, España.

Correspondence

 

The suspected radiological diagnosis was of intraosseous hemangioma of the mandible in view of the images described, and because the vascular channel was nearby and enlarged in comparison with the contralateral side.

A preoperative study was carried out that did not contraindicate surgical intervention. Under general anesthesia and by means of an intraoral approach involving an incision into the back of the lower right vestibule once the right mental nerve had been identified and preserved, the tumor-like mass was removed after ligation of the feeding artery with a silk suture and while applying Tissucol® y Surgicel® to the surgical bed. The postoperative period was complicationfree. The pathology report of the specimen was of cavernous hemangioma of the mandible. The patient has been followed for 13 months on an outpatient basis and a good outcome has been achieved. Nine months after the intervention she had a follow-up CAT scan that showed the surgical defect in the process of regenerating, and there were no signs of local recurrence.

 

Discussion

Intraosseous hemangiomas are benign lesions, due to the proliferation of blood vessels, and they are very unusual. They account for 0.2% of all bone neoplasms, and they are generally located in the vertebrae and the skull1 followed by the mandible and the maxilla, and they are more commonly found at the back.2,3

Their etiology is unknown but it is thought that some are real neoplasms and that others have a traumatic origin.2 They are twice as common in men as in women, and the peak incidence is in the second decade of life.2,4

Most are incidental radiographic findings5 and there are no clinical symptoms,2-4 although it is true that on some occasions erosion and resorption of teeth is produced5 (the teeth affected become mobile). There is facial disfiguration from the asymmetrical expansion as a result of the slow growth of the maxilla or mandible, pain or paresthesias. Also described are pulsating hemangiomas on auscultation or on palpation of the thin bone sections, together with spontaneous bleeding of the gums around the teeth in that area. There can even be catastrophic bleeding after extraction of a tooth6 associated with a hemangioma or on carrying out a biopsy.7 In order to avoid these accidents the suspected diagnosis is very important together with imaging studies.24

Bearing in mind that biopsies are contraindicated due to the high risk of bleeding, the suspected diagnosis is carried out by means of radiological and clinical studies, although it should be kept in mind that the radiologic presentation is very varied2-4 (up to 12 different appearances have been described)8 and unspecific. The enlargement of the lower dental nerve is suggestive of a lesion with an avascular origin. 8 There are authors that maintain that in half the cases a sclerotic margin can be observed.9 Multilocular radiolucencies can appear with a similar appearance to soap bubbles. It may also appear as a rounded radiolucent lesion in which a trabecular bone can be seen in the center. A cysticlike radiolucent appearance is less common.10

The differential diagnosis should be made with other entities with a compatible radiological appearance, such as the residual dental cyst, odontogenic keratocyst, aneurismatic bone cyst, ameloblastoma, odontogenic myxoma, giant cell granuloma, and other similar pathologies.10

Before treatment, evaluating three factors is very important: size, location, and vascular supply. It is as from this point that the conflict arises, as different therapeutic procedures have been described: surgery, ligation of the external carotid artery, radiotherapy,11 inoculation of sclerosing agents,12 cryotherapy, and embolization.13 On some occasions carrying out an angiography before deciding upon one of these options can be very useful.

Although the use of isolated embolization13 has been described as successful (that may have to be repeated for complete devascularization), the best treatment may consist in carrying out a superselective angiography with embolization, and with surgical excision should it be necessary, although there should not be a gap of more than two weeks between these in order to avoid collateral vessel formation. 14 Embolization is not an innocuous process, and among the complications described is the breaking off of pieces of atheroma, broken catheters, broken vessels,15 hemiplegia, blindness, facial paralysis,16 allergic reactions. The risk of avascular necrosis of the mandible has been documented after embolization as a therapeutic option followed by surgery.17 It would be of interest if during the surgery controlled hypotension could be carried out by the anesthetist, and regulated hypothermia maintained, in order to reduce blood loss.18

Generally the arteries supplying the hemangioma proceed principally, but not always, from the external carotid artery. Despite this, it is important to take into account that the ligation of the external carotid artery will lead to the opening of collateral channels from the contralateral external carotid artery5,16,19 (that can turn out to be inaccessible for embolization with catheters), or in the ipsilateral internal carotid artery5 (that could lead to the lesion increasing, due to increased pressure). In other words, it cannot be assumed that the ligation of the E.C.A will control acute bleeding or that adequate devascularization will be produced of the vascular lesion.20-22

Although the use of injected sclerosing agents has been described, they can turn out to be very dangerous due to the proximity of the intracranial vascular system.12 Radiotherapy can be unsuccessful given the level of maturity of the cells in the malformation, the risk of sarcoma development, the production of osteoradionecrosis delaying osseous and dental development, and possible surgical intervention at a later date may be complicated.23 The definitive diagnosis is through the pathologic analysis of the tumor. Most are of the cavernous type, in which the dilated vascular spaces with fine walls are surrounded by benign endothelial cells. It is also possible for there to be numerous vascular channels and others of the size of a capillary, although a mixture of both subtypes is possible.2-4

Despite the fact that we are dealing with an entity with benign characteristics, prompt treatment is recommended in order to avoid possible complications as a result of local growth, that could even lead to a somber prognosis as a result of catastrophic bleeding. With correct treatment the prognosis of these lesions is good, with bone regenerating in the affected area during the following months. Recurrence is rare.

 

Conclusions

The intraosseous hemangioma of the mandible is an unusual pathology, frequently diagnosed as a result of incidental findings following radiological tests for other reasons. There tend to be no symptoms, unless allowed to develop, and the prognosis is good.

With regard to treatment there are various possibilities, although surgical treatment as an option, with previous embolization, is considered as very valid. Carrying out a presurgical superselective angiography can be useful.

 

 

Correspondence:
Ramon Luaces Rey
Centro Hospitalario Universitario Juan Canalejo
Xubias de Ariba 84, 15006 A Coruña, España.
Email. rluarey@canalejo.org

 

 

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