|Year : 2015 | Volume
| Issue : 3 | Page : 83-87
Cavernous hemangioma of mandible: A rare case report
Neeraj Kumar Dhiman
Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh, India
|Date of Web Publication||27-Nov-2015|
Dr. Neeraj Kumar Dhiman
Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
Intraosseous vascular lesions are rare conditions, comprising only 0.5-1% of all intraosseous tumors. Found mainly in the second decade of life, especially in female. Vertebral column and skull are the most common sites; nevertheless, the mandible is a quite rare location. Hemangiomas are benign vasoformative neoplasms of endothelial origin. However, the origin of central hemangioma is debatable. Some authors believe that it is a true neoplasm, whereas others state it as a hamartomatous lesion. The clinical and radiographic presentation is relatively nonspecific; therefore, a proper diagnosis has to be made. Clinically, the patient may be completely symptom-free or may present pulsatile bleeding, slow growing bluish mass, mobile teeth, and deranged dentition, early dental exfoliation and discomfort in normal life. Cavernous hemangioma produce dilemma in diagnosis with neoplasms such as ameloblastoma, cystic lesions such as residual cyst, keratocyst, osteosarcoma, central giant cell granuloma, multiple myeloma, and fibro-osseous lesions such as fibrous dysplasia. Among various treatment modalities, surgery is frequently used. A case report of 14-year-old male patient with cavernous hemangioma of right mandibular body with vague clinical features, but a characteristic radiographic and histological picture is presented here.
Keywords: Hemangioma, intraosseous, mandible, vascular malformation
|How to cite this article:|
Dhiman NK. Cavernous hemangioma of mandible: A rare case report. J Oral Maxillofac Radiol 2015;3:83-7
| Introduction|| |
Hemangioma is a benign vasoformative neoplasm of endothelial origin. , Its origin is debatable. Shira and Guernsey  believe that it is a true benign neoplasm as a result of initial endothelial proliferation, which then differentiates into blood vessels. Others state that it is a hamartoma resulting from the proliferation of mesoderm that undergoes endothelial differentiation and subsequently, is canalized and vascularized.  Intraosseous hemangioma is a quite rare condition, comprising <1% of all intraosseous tumors. It mainly occurs in the vertebral column and skull. Mandible is a very infrequent location although possible. The female:male ratio is 2:1 and the peak of incidence is between the second and fifth decades of life. ,
Usually, it is symptomless but may present signs and symptoms such as a slow growing bluish mass, pulsatile sensation, deranged dentition, recurrent bleeding due to any trauma, mobile teeth, and discomfort in normal life. Panoramic radiograph, computed tomography (CT) scan, magnetic resonance imaging (MRI), and CT angiography are the most useful radiographic tools. CT scan allows clear visualization of cortical involvement, while MRI and CT angiography shows blood flow from feeding vessel, if present, as well as the relationship with surrounding soft tissues. 
Central hemangiomas of bone arise from vessels within the marrow spaces and may comprise arterial and venous vessels. Microscopically, engorged vascular sinuses are present with an endothelial lining supported by a connective tissue stroma interspersed by bony trabeculae, which are usually arranged at right angles to the surface.  Radiographically, honeycombed or sunburst appearance with spindles radiating toward the periphery is present which is a differentiating feature of hemangioma from ameloblastoma.
Central hemangioma is a great mimicker as it resembles to central giant cell granuloma, fibrous dysplasia, multiple myeloma, osteosarcoma, ameloblastoma, and keratocyst radiographically. Patients are at high risk of bleeding due to any traumatism or extraction attempt. Therefore, the patient's history, radiographs, and other investigations play an important role in making final diagnosis and treatment plan. In this article, a case of a patient diagnosed as a cavernous hemangioma of right mandibular body is reported.
| Case Report|| |
A 14-year-old male child reported with the chief complaint of swelling on the right side of the face over the previous 1-year. Patient was apparently asymptomatic before he observed this swelling 1-year back associated with mild pain. The swelling started small, reached its present size in 6 months with gradual decrease in pain intensity, and had been the same ever since. No history of paresthesia, spontaneous bleeding, or epistaxis was given.
On extra-oral examination, facial asymmetry was evident on the right side. The swelling was approximately 4 cm × 3 cm in size, present over the right mandibular body with a smooth surface and diffuse margins. Palpation revealed a bony, hard, nontender swelling with free skin and normal temperature in relation to the lateral aspect and lower border from the angle of mouth to the mandibular angle on right side. Intraoral examination exhibited obliteration of the buccal vestibule extending from the mandibular right first premolar to the retromolar region with redness in the mucosa. The dentition shows deflection of the crowns of both premolars and first molar, second molar was almost lingually inclined and third molar was embedded in soft tissue [Figure 1]. Teeth present in the involved area were vital and firm. The canine was present in labial relation to central incisors. All the other findings were noncontributory.
A panoramic radiograph discloses multiple coarse trabeculations and increased radiodensity extending from the mandibular right first premolar to the mandibular angle and some part of ramus [Figure 2]. Differential diagnosis included solitary bone cyst, ameloblastoma, myxoma, giant cell lesion, and bone hemangioma. The CT scan findings revealed an ill-defined, heterogenous, multiloculated, and multiseptated osteolytic lesion of size 6.2 cm × 2.7 cm × 4.8 cm (AP × Tr × Longitudinal) involving alveolar process of right hemi-mandible, angle and proximal ramus having honeycomb appearance [Figure 3]. Multiple area of cortical breaks and thinning of the outer cortex show a characteristic sunburst appearance of the periosteum with spindle radiating from the center to periphery of lesion suggestive of cavernous hemangioma of the mandible [Figure 4].
|Figure 3: Three-dimensional computed tomography showing extension and involvement of outer mandibular cortex|
Click here to view
|Figure 4: Axial computed tomography view showing involvement of mandible and periosteal reaction|
Click here to view
Fine-needle aspiration cytology was performed before biopsy to confirm the diagnosis. It revealed a little hematic material with no apparent cellularity. During biopsy bleeding from the lesion was manageable, but creates a dought of vascular lesion. After closure, the tissue was sent for histopathological examination. Microscopic examination of the specimen showed lamellar bone with closely packed, thin-walled dilated blood vessels lined by flattened endothelial cells [Figure 5]. There were no atipias or mitotic figures. The histopathologic diagnosis was cavernous hemangioma of the mandible.
Arteriography was done which does not show any feeding vessel to the lesion [Figure 6]. Wide surgical excision of the lesion including healthy margins performed. A reconstruction locking plate (spacer) was used to bridge the defect and to maintain the space for bone grafting at a later stage [Figure 7]. The mandibular fragment was sent for histopathological diagnosis, which again confirmed the presurgical diagnosis of cavernous hemangioma. The patient has been on follow-up since 6 weeks postsurgery, the wound is healing uneventfully, and the reconstruction of the mandibular defect via grafting is being planned.
|Figure 6: Computed tomography angiography revealing absence of any feeding vessel to the lesion|
Click here to view
|Figure 7: Intraoperative picture showing bridging of defect with reconstruction plate|
Click here to view
| Discussion|| |
The most frequent location of hemangioma is the molar-premolar region.  Pathogenesis is still debatable  and several theories are postulated. Some authors describe hemangiomas as congenital lesions whereas others believe that the inferior dental canal is the origin of the lesion, based on its widening in the majority of these patients. , The initial diagnosis is usually complicated because of the absence of symptoms and the unspecific radiological findings. , Radiographically, a differential diagnosis of ameloblastoma, cavernous hemangioma, giant cell lesion, cyst, and myxoma could be made due to the characteristic sunburst appearance. ,, Clinical history, examination findings, radiographs, and scanning examination illustrates many features which show characteristic of central hemangioma.
The CT-scan allows clear visualization of cortical involvement, , and is also useful to define the extension of the hemangioma and its relationship with surrounding soft tissues.  The classical feature is the "polka-dot" appearance with cortical expansion. Honeycombed appearance and periostic reaction are extremely rare presentations. , A CT angiography is needed to rule out any feeding vessel. Preoperative arteriography is usually unnecessary because a vascular flow cannot be identified in the majority of the cases. , Nevertheless, it should be performed together with a presurgical embolization in big lesions to minimize the surgical bleeding. ,
There are two types of hemangioma: Peripheral and central. Peripheral hemangioma is originated in the periostic vessels that grow into the medullar bone, while central hemangioma is originated into the medullar bone and grow toward the cortical bone. Histologically, hemangioma can be divided into three groups: Cavernous, is the most frequent one and is located into the mandible,  capillary and mixed.
Treatment is indicated only in some conditions: Esthetic disfigurement, repetitive bleeding, and palpable mass. Clinical observation is only indicated in two conditions: Asymptomatic patients or minimal facial deformity.  Therapeutic alternatives include: Surgery, curettage, radiotherapy, and embolization.  Systemic or intralesional corticosteroids can also be used along with angiogenesis inhibitors, such interferon in selected cases.  Intralesional injections of sclerosing agents would not have been effective because of the lesion's bony nature.
Surgical excision with reconstruction of mandible remains the preferred treatment. Prognosis after complete excision is excellent and recurrence is usually rare. , Simple curettage may lead to an uncontrollable bleeding as well as an incomplete excision of the lesion.  Radiotherapy was not the treatment of choice, considering the age of the patient and the retarding effects of radiation on oral and perioral tissues. Percutaneous embolization has been defended by several authors, although technical risks are greater than benefits obtained. 
| Conclusion|| |
The elective treatment should be a wide excision of the lesion including healthy surrounding bone, as well as ligature of the nutritional vessels, if present. , The identification of feeding vessels is very important for the management of vascular lesions. In cavernous hemangioma of bone, though, conventional radiographs give us adequate information regarding the extent and nature of the lesion, specialized radiographic techniques such as CT angiography would help in detecting the inner component of the lesion and identification of the feeder vessels if present. Because of the serious consequences, hemangiomas must always be considered in the differential diagnosis and proper precautions must be taken in establishing the final diagnosis before any surgical treatment is undertaken.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Alves S, Junqueira JL, de Oliveira EM, Pieri SS, de Magalhães MH, Dos Santos Pinto D Jr, et al.
Condylar hemangioma: Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:e23-7.
Williams HJ, Wake MJ, John PR. Intraosseous haemangioma of the mandible: A case report. Pediatr Radiol 2002;32:605-8.
Shira RB, Guernsey LH. Central cavernous hemangioma of the mandible: Report of case. J Oral Surg 1965;23:636-42.
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.
Cheng NC, Lai DM, Hsie MH, Liao SL, Chen YB. Intraosseous hemangiomas of the facial bone. Plast Reconstr Surg 2006; 117:2366-72.
Fernández LR, Luberti RF, Domínguez FV. Radiographic features of osseous hemangioma in the maxillo-facial region. Bibliographic review and case report. Med Oral 2003;8:166-77.
Marwah N, Agnihotri A, Dutta S. Central hemangioma: An overview and case report. Pediatr Dent 2006;28:460-6.
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.
Mohnac AM, Smith JR. Central hemangioma of the mandible: Report of case. J Oral Surg 1967;25:455-9.
Jaffe HL. Tumors and tumorous conditions of the bones and joints. Philadelphia, PA: Lea & Febiger Publishers; 1953. p. 236-7.
Zlotogorski A, Buchner A, Kaffe I, Schwartz-Arad D. Radiological features of central haemangioma of the jaws. Dentomaxillofac Radiol 2005;34:292-6.
Perugini M, Renzi G, Gasparini G, Cerulli G, Becelli R. Intraosseous hemangioma of the maxillofacial district: Clinical analysis and surgical treatment in 10 consecutive patients. J Craniofac Surg 2004;15:980-5.
Behnia H, Ghodoosi I, Motamedi MH, Khojasteh A, Masjedi A. Treatment of arteriovenous malformations: Assessment of 2 techniques - Transmandibular curettage versus resection and immediate replantation. J Oral Maxillofac Surg 2008;66:2557-65.
Shum JW, Clayman L. Resection and immediate reconstruction of a pediatric vascular malformation in the mandible: Case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:517-24.
Jayakumar PN, Desai SV, Kovoor JM, Vasudev MK. Percutaneous embolization of mandibular hemangioma: A case report. J Oral Maxillofac Surg 2002;60:945-8.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]