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

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.

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 varied^2-4 (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 artery^5,16,19 (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.

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.

References

1. Faerber T, Hiatt WR. Haemagioma of the frontal bone. J Oral Maxillofac Surg 1991;49:1018.        [ Links ]

2. Yih WI, Ma GS, Merril RG, y cols. Central Hemangioma of the jaws. J Oral Maxillofac Surg 1989;47:1154.        [ Links ]

3. Princ, Gasnier F, Naman N, y cols. Angiomes de Maxillaires. Rev Stomatol Chir Maxillofac 1985;86:295.        [ Links ]

4. Loche MJ, Cock H, Friedlander A. Central arteriovenous malformation on the maxillofacial skeleton. J Oral Maxillofac Surg 1991;49:759.        [ Links ]

5. Guibert-Trainer F, Piton J, Riche MC, Merland JJ, Caille JM. Vascular malformation of the mandibuke (intraosseus haemengioma). The importance of preoperative embolization . A study of nine cases. Eur J Radiol 1982;2:257-72.        [ Links ]

6. Taylor BG, Etheridge SN. Hemangiomas of the mandibule and the maxilla presenting as surgical emergencies. Am J Surg 1964;108:574.        [ Links ]

7. Lamberg MA, Tasanen A, Jaaskelainen J. Fatality fron central hemangioma of the mandibule. J Oral Surg 1979;37:578.        [ Links ]

8. Worth HM, Stoneman DW. Radiology of vascular abnormalities in and about the jaws. Dent Radiography Photography 1979;52:1-23.        [ Links ]

9. Langlais RP, Langland OE, Nortje CJ. Multilocular radiolucencies. Diagnostic Imaging of the Jaws. Malern: Williams and Wilkins 1995;327- 384.        [ Links ]

10. Beziat JL, Marcelino JP, Bascoulergue Y, Vitrey D. Central vascular malformation of the mandibule: a case report. J Oral Maxillofac Surg 1997;55:415-9.        [ Links ]

11. Laws IM. Pulsating Hemangima of the jaws. Br J Oral Surg 1968;5:223.        [ Links ]

12. Clin DC,. Treatment of maxillary hemangioma with a sclerosing agent. Oral Surg Oral Med Oral Pathol 1983;55:247.        [ Links ]

13. Bien S, Garcia-Cervignon E, Laurent A , y cols. Guerison par traitment endovasculaire transcutane dùn angiome de la mandibule et des parties molles adjacentes. Rev Stomatol Chir Maxillofac 1988;89:148.        [ Links ]

14. Hupp JR. Superselective angiography with distal subtraction and embolization of a maxillary haemangioma in a patient?        [ Links ]

15. McKenna SJ, Roddy SC. Delayed Management of a Mandibular Vascular Malformation. J Oral Maxillofacial Surg 1989;47:517-22. 16. Braun IF, Levy S. The use of transarterial microembolization in the management of hemangiomas of the perioral region. J Oral Maxillofac Surg 1985;43:239.        [ Links ]         [ Links ]

17. Gallagher DM, Hilley D, Epker BN. Surgical treatment of an arteriovenous malformation of the mandibule in a child. A case report. Maxillofac Surg 1983;11:279.        [ Links ]

18. Mulliken JB, Murray JE, Castaneda AR, et al. Management of vascular malformations of the face using total circulatory arrest. Surg Gynecol Obstet 1978;146:168.        [ Links ]

19. Yeoman CM. Management of hemangioma involving facial, mandibular and pharyngeal structures. Br J Oral Maxillofac Surg 1987;825:195.        [ Links ]

20. Schindel J, Matz S, Edlan A, y cols. Central cavernous hemangioma of the jaws. J Oral Surg 1978;36:803.        [ Links ]

21. Spatz S, Kaltman S, Fraber S. Vascular malformation: Report of a case with eight year follow-up. J Oral Surg 1985;43:381.        [ Links ]

22. Selfe RW, Sherman M, Miller TF. Arteriovenous malformations of the mandibule. Otolaryngology 1978;86:659.        [ Links ]

23. Perriman A, Uthman A, Kuzair KY. Central hemangioma of the jaws. Oral Surg 1974;37:502.        [ Links ]

24. Drage N, Whaites E, Hussain K. Haemangioma of the body of the mandibule : a case report. Br J Oral Maxillofac Surg 2003;41:112-4.        [ Links ]