|Year : 2009 | Volume
| Issue : 1 | Page : 42-45
Central hemangioma of mandible presenting as massive radiolucency
Simi Thankappan, Valsa Thomas, Nileena R Kumar, KP Sharafudeen, Sherin Nair
Department of Oral Medicine and Radiology, Government Dental College, Calicut-673 008, Kerala, India
|Date of Web Publication||14-Nov-2009|
Department of Oral Medicine and Radiology, Government Dental College, Calicut-673 008
Source of Support: None, Conflict of Interest: None
| Abstract|| |
A hemangioma is a proliferation of blood vessels that creates a mass resembling a neoplasm. Some regard it as a true neoplasm while others state that it is a hamartoma resulting from proliferation of mesodermal cells that undergo endothelial differentiation and subsequently are canalized and vascularized. A central hemangioma rarely develops in the jaws. This case report deals with a low flow hemangioma in the ramus of the mandible, which despite its large size proved to be innocuous and lacked most of the characteristics of a conventional central hemangioma.
Keywords: Angiography, central hemangioma, honeycomb appearance, ultrasonography, curettage
|How to cite this article:|
Thankappan S, Thomas V, Kumar NR, Sharafudeen K P, Nair S. Central hemangioma of mandible presenting as massive radiolucency. J Indian Acad Oral Med Radiol 2009;21:42-5
|How to cite this URL:|
Thankappan S, Thomas V, Kumar NR, Sharafudeen K P, Nair S. Central hemangioma of mandible presenting as massive radiolucency. J Indian Acad Oral Med Radiol [serial online] 2009 [cited 2019 Apr 20];21:42-5. Available from: http://www.jiaomr.in/text.asp?2009/21/1/42/57778
| Introduction|| |
The central hemangioma is a vascular neoplasm of endothelial origin  and rarely develops in the jaws, , and accounts for approximately 1% of all primary bone tumors.  It is frequently found in the vertebrae and the skull. Occurrence in the mandible is very rare.  The following case report shows a different clinical, radiographic and treatment picture regarding central hemangioma.
| Case Report|| |
A 16-year-old male was referred to the department of oral medicine and radiology from a private dental practitioner for the evaluation of a cystic radiolucency in relation to the lower left second molar which was extracted. The patient's complaint was salty fluid coming out from the socket of the tooth which was extracted two weeks previously due to caries.
When asked about the swelling on the left side of the face, he revealed that the swelling was present since childhood, first noticed at about 1 year of age and since it persisted with slight increase in size, they came to the dental college at the age of 5 years. Radiographs were taken at that time. They were informed that there was only slight thickening in the bone and if it increased in size, it should be removed. Since there was no further increase in the size of swelling as perceived by the patient, they didn't come for any treatment. About one year back, frequent toothache started in same region and he consulted the dentist. A small radiograph was taken before extraction. They were informed that the external swelling was due to a cyst associated with decayed tooth, and that it would regress after extraction. After extraction of the second molar, salty fluid started coming out from the site. Besides, the swelling became more obvious. So they consulted the same dentist and the patient was referred to the dental college for expert management.
On presentation, he had a large swelling on the left ramus of the mandible with no history of pain, bleeding or paresthesias [Figure 1]. The skin over the lesion appeared normal. He had decreased mouth opening of 25 mm and a history of progressive decrease in mouth opening due to locking. Intra-orally, retromolar region posterior to socket of 37 was obliterated and it was about 1.5 Χ 1.5 cm with hard bony consistency with mild degree of fluctuance of 5 mm area just posterior to 37 socket. Mild compressibility was also elicited in the same area without any pulsations. There was no evidence of bleeding from the gingival sulcus.
Radiographs were taken and the panoramic radiograph showed a large well-defined unilocular radiolucency of size 7 Χ 4 cm, involving the ramus, angle and coronoid and condyle of left mandible [Figure 2]. There was expansion of anterior and posterior borders of ramus throughout the entire height of ramus with postero-inferior expansion of angle and slight obliteration of sigmoid notch. The cortical plate was thinned out with no perforation visible in radiograph. There was overlapping of hyoid bone in inferior part and airway shadows in upper and middle part of the lesion. Mandibular canal was displaced to inferior part about 1 cm from the angle of the mandible. There was developmental missing of third molar crypt. On aspiration blood tinged dirty fluid was obtained.
After correlating the history, clinical and radiographic presentation, a differential diagnosis of primordial cyst, dentigerous cyst, ameloblastoma or keratinizing cystic odontogenic tumor (KCOT) or overlapping of two or more lesions on childhood lesions such as aneurysmal bone cyst with fibro-osseous lesion was made. Blood and serum chemistries were normal. An ultrasonography of the lesion was done which gave vascular lesion as the diagnosis. Plain angiography confirmed it as a low flow vascular lesion. A biopsy was done and there was only minimal bleeding, and the lesion was histopathologically diagnosed as capillary hemangioma [Figure 3].
Due to the patient's reluctance for CT angiography because of the expense, it was not done. The lesion was opened surgically and because of less bleeding and size of the lesion, the solid components in the lesion were curetted and marsupialized. The lesion is healing uneventfully now and no complications have been reported in the 6-month follow-up period [Figure 4] and [Figure 5].
| Discussion|| |
Central hemangioma can be either asymptomatic/symptomatic, and can show signs and symptoms like discomfort, oozing or pulsatile bleeding from the gingiva around the teeth in the region of the lesion, bluish discoloration of gingiva and mobile teeth. Highly expansile lesions cause sensation of pulsation, audible bruits on extension into the soft tissue and blanching on pressure. Cases of spontaneous bleeding and paresthesia have also been reported.  But in this patient, no such findings were present and the only symptom was the innocuous swelling which persisted over a period of 15 years.
In a review of 84 cases of central hemangiomas by Zlotogorski et al., the female to male ratio was 1.3:1  with the peak incidence in the second decade.  Regarding the site, 77% cases were located in the mandible and 23% in the maxilla. A majority of the lesions occurred in the posterior region of mandible,  mainly in the molar-premolar region,  but condylar tumors have also been reported in the literature. 
The radiographic appearance of central hemangioma is not pathognomonic and can simulate many other bone lesions. Vascular lesions in general are also called "great imitators' because of the marked variations in appearance.  Most commonly, lesions show a multilocular radiolucency with honeycomb or soap-bubble appearance. Lesions may also present as a unilocular radiolucency. , According to Worth, there is an additional pattern, in which the trabeculae resemble the spokes of a wheel, radiating from the centre of the lesion. 
In the review by Zlotogorski et al., 96% of cases were radiolucent lesions. Large and extensive lesions that represented more than one radiographic pattern were classified according to the predominant radiographic feature. The lesions were multilocular (66%), unilocular in 33%, and sunray pattern (not loculated) in only one case (1%). Of the multilocular lesions, 30% presented a honeycomb pattern, 24% a soap-bubble pattern, and 7% a spoke-like pattern. Borders were diffuse in a majority of the cases. In 70%, the size was more than 2 cm. Tooth resorption was described in 23% cases and tooth displacement in 16% cases. Radiological signs of sinus involvement were found in 35% of the maxillary lesions. In 15% of the mandibular lesions, there was inferior alveolar nerve canal involvement, usually manifested as an enlargement of the canal width. 
Radiographic differential diagnosis includes osteosarcoma, fibrous dysplasia, central giant cell granuloma, ameloblastoma, odontogenic myxoma, multiple myeloma, dentigerous cyst and aneurysmal bone cyst. 
The central hemangioma may be clinically and radiographically indistinguishable from other vascular conditions known as central arteriovenous fistula or aneurysm. Angiography has proved to be useful as a diagnostic tool in demonstrating the presence of a vascular lesion, to delineate its boundaries, also in differentiating arteriovenous fistula and central hemangioma. 
The CT scan allows clear visualization of cortical involvement, extension of the hemangioma, and the relationship with surrounding soft tissues. Some authors have described it as having "polka-dot" appearance with cortical expansion.  Biopsy is formally contraindicated because of the high risk of bleeding. 
Management of central hemangioma in the mandible has been done using various modalities like surgery, radiotherapy, curettage and embolization.  The difficulty in these lesions is the abundance of vascularity. Of the various therapeutic modalities, surgery is the most frequently used. Intralesional injections of sclerosing agents have been reported successful in a case.  Complete curettage is advocated as the treatment in children. The risk of bleeding can be reduced by ligature of the inferior alveolar artery during surgery.  Simple curettage was done in this case, and the lesion has been regressing without any complications.
| Summary|| |
Histopathological surprise of a hemangioma can be dangerous during the initial incision biopsy and can prove fatal to the patient. In this patient, central hemangioma was not included in the differential diagnosis because of the rarity of the lesion. But since it was a low flow lesion and did not show characteristic features, it was treated by curettage, which is mostly done in case of aneurysmal bone cyst. The present study demonstrated that this lesion should be considered in the differential diagnosis of any multilocular or unilocular radiolucent lesion of the jaws, especially when the mandible is involved. It is very obvious from the preceding features that the most illustrious attribute of hemangioma radiographically is the lack of a characteristic picture.
| Acknowledgment|| |
Dr. Nileena R. Kumar, MDS Oral Medicine and Radiology, Senior Lecturer, Department of Oral Medicine and Radiology, Government Dental College, Calicut-673008.
| References|| |
|1.||Gσmez Oliveira G, Garcνa-Rozado A, Luaces Rey R. Intraosseous mandibular haemangioma: A case report and review of the literature. Med Oral Patol Oral Cir Bucal 2008;13:E496-8. |
|2.||Nagpal A, Suhas S, Ahsan A, Pai KM, Rao NN. Central haemangioma: Variance in radiographic appearance. Dentomaxillofac Radiol 2005;34:120-5. |
|3.||Zlotogorski A, Buchner A, Kaffe I, Schwartz -Arad D. Radiological features of central haemangioma of the jaws. Dentomaxillofac Radiol 2005;34:292-6. |
|4.||Hayashi T, Ito J, Kato T, Hinoki A, Taira S, Saku T. Intracortical hemangioma of the mandible. Dentomaxillofac Radiol 1999; 28:127-9. |
|5.||Drage NA, Whaites EJ, Hussain K. Haemangioma of the body of the mandible: A case report. Br J Oral Maxillofac Surg 2003;41:112-4. |
|6.||Wood NK, Goaz PW, Kallal RH. Multilocular radiolucencies. In: Wood NK, Goaz PW, editors. Differential diagnosis of oral and maxillofacial lesions. 5 th ed. Missouri: Elsevier; 1997. p. 348-9. |
|7.||Worth HM. In: Benign Tumors of the Jaws; Principles and Practice of Radiologic Interpretation. USA: Year Book Medical Publishers Inc; 1963. p. 522-7. |
|8.||Wang X, Wang C, Fu F, Gao Q. Diagnosis and treatment of central hemangioma of mandible. Hua Xi Kou Qiang Yi Xue Za Zhi 1999;17:244-7. |
|9.||Williams HJ, Wake MJ, John PR. Intraosseous haemangioma of the mandible: A case report. Pediatr Radiol 2002;32:605-8. |
|10.||Kaneko R, Tohnai I, Ueda M, Negoro M, Yoshida J, Yamada Y. Curative treatment of central hemangioma in the mandible by direct puncture and embolisation with n-butyl-cyanoacrylate (NBCA). Oral Oncol 2001;37:605-8. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]