|Year : 2016 | Volume
| Issue : 2 | Page : 211-214
Differential diagnosis of central hemangioma of maxilla: A rare case report
Vennila Parameswaran, Chandrasekhar Thirumoorthy, Senthil Kumar Marappa Gouder Sathyamoorthy, Priyadharshini Ramakrishnan
Department of Oral Medicine and Radiology, Sri Ramakrishna Dental College and Hospital, Coimbatore, Tamil Nadu, India
|Date of Submission||01-Jun-2015|
|Date of Acceptance||21-Nov-2016|
|Date of Web Publication||02-Dec-2016|
Department of Oral Medicine and Radiology, Sri Ramakrishna Dental College and Hospital, Coimbatore, Tamil Nadu
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Central hemangioma of the maxilla is a rare entity. Although the mucosal and soft tissue lesions are readily suspected due to their clinical appearance, the intrabony lesions may be difficult to distinguish visually. Early diagnosis of central hemangioma with appropriate and efficient diagnostic modality can help in the effective and more conservative management of the condition, eliminating the risk of hemorrhage following biopsy. Here, we present a case of central hemangioma of the maxilla emphasizing on various radiographic differential diagnosis with advanced imaging techniques.
Keywords: Central hemangioma, differential diagnosis, maxilla
|How to cite this article:|
Parameswaran V, Thirumoorthy C, Sathyamoorthy SM, Ramakrishnan P. Differential diagnosis of central hemangioma of maxilla: A rare case report. J Indian Acad Oral Med Radiol 2016;28:211-4
|How to cite this URL:|
Parameswaran V, Thirumoorthy C, Sathyamoorthy SM, Ramakrishnan P. Differential diagnosis of central hemangioma of maxilla: A rare case report. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2021 Apr 13];28:211-4. Available from: https://www.jiaomr.in/text.asp?2016/28/2/211/195142
| Introduction|| |
Hemangioma is a true benign neoplasm of initial endothelial proliferation that differentiates into blood vessels.  Some authors state that it is a hamartoma resulting from the proliferation of mesoderm that undergoes endothelial differentiation and, subsequently, is canalized and vascularized.  Hemangiomas can occur anywhere in the body but are most frequently noticed in the skin and subcutaneous tissues. Central hemangioma (intraosseous) is most often found in the vertebrae and skull, rarely in jaws, with mandible being involved twice as often as the maxilla with a male:female ratio of 1:3 and a peak incidence in the second decade of life. ,
| Case Report|| |
A 29-year-old female patient reported to the Department of Oral Medicine Radiology and Diagnosis, with the chief complaint of recurrent swelling in the right upper back tooth region for the past 4 years. The first incidence of swelling was observed in 2008. The swelling was gradual in onset and slowly increased in size without episodes of pain. The patient underwent a surgical excision of the swelling, however, she reported back with the same recurrent swelling to our department in 2015. The patient's past medical history and personal history were not contributory.
On extraoral examination, the patient presented with a solitary diffuse swelling of the right middle third of face causing gross facial asymmetry [Figure 1], which was hard in consistency, nontender, and without cervical lymphadenopathy. Intraoral examination revealed a diffuse smooth-surfaced swelling in the palatal mucosa, which was firm in consistency, pulsatile in nature with tenderness on palpation extending from 13 to 16 tooth region with mild cortical expansion [Figure 2]. On hard tissue examination, 16 was missing. By correlating the pulsatile and recurrent nature of the swelling, a provisional diagnosis of central hemangioma was made.
|Figure 1: Extraoral photograph showing diffuse swelling of the right middle-third of the face|
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|Figure 2: Intraoral photograph showing diffuse swelling in the right palatal mucosa with mild cortical expansion|
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Occlusal radiograph [Figure 3] of the involved region showed an increased radiopacity, with panoramic view [Figure 4] revealing a well-defined lytic lesion at the right maxilla related to the root of the premolar and canine teeth, elevating the floor of the right maxillary sinus, both of which mimicked ground glass appearance. Contrast computed tomography (CT) scan showed an expansile lytic lesion involving the right maxilla and palate, elevating the floor of the maxillary sinus and nasal cavity with dehiscence of bone in relation to the oral cavity. Trabeculations and thin bony spicules were seen within the margins of the lesion, suggesting a sunburst appearance [Figure 5]. The lesion showed evidence of flow voids on T1 weighted [Figure 6] and T2 weighted images with diffuse enhancement following contrast. No soft tissue invasion was noted. [Figure 7]. On fine-needle aspiration cytology (FNAC), there was a spurt of blood from the swelling. Because biopsy carried a greater risk of hemorrhage, the patient was referred to a higher centre for further effective management of the condition.
|Figure 3: Occlusal radiography presenting increased radiopacity of the involved region|
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|Figure 4: Panoramic view presenting a well-defined lytic lesion in the right maxilla|
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|Figure 5: Contrast computed tomography scan showed an expansile lytic lesion involving the right maxilla and palate elevating the floor of the maxillary sinus with diffuse enhancement on contrast|
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|Figure 6: An axial T1 weighted magnetic resonance image showing low signal expansile lesion in the right maxilla|
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|Figure 7: An axial T2 weighted MR image showing low to intermediate signal expansile lesion in the right maxilla|
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| Discussion|| |
Central hemangiomas are probably congenital, developmental or traumatic in origin, usually asymptomatic and locally destructive occasionally. It presents as a slow-growing painless pulsatile swelling of the bone, which may or may not cause facial asymmetry, ,, which correlated with our case. Authors have also reported features like discomfort, oozing or pulsatile bleeding from the gingiva of teeth in the region of the lesion, mobile teeth, accelerated exfoliation of teeth and audible bruits in lesions extending to soft tissues.  Despite the benign nature of the lesion, paraesthesia in the region is not uncommon.
The central haemangioma of bone has been referred to as "great imitator" with different radiographic variations.  For example:
A multilocular appearance can be detected in 50% of the cases. , Course linear trabeculae that radiate from the centre of the lesion with small angular locules of varying shape are seen, with a round outline. 
A cyst-like radiolucency with an empty cavity and sometimes a hyperostotic border may also be observed. 
Many authors have described the classical sunburst or sunray appearance, which is caused by coarse trabeculae perpendicular to the bony surface, a feature that was noted in our case.  Resorption of roots of the involved teeth occurs with some frequency, and calcifications appearing as radiopaque rings are occasionally seen.  Therefore, a radiographic differential diagnosis of (1) ameloblastoma; (2) central giant cell granuloma; (3) odontogenic myxoma; (4) osteosarcoma; (5) fibrous dysplasia; and (6) aneurysmal bone cyst ,, were considered.
The presence of a missing tooth, age and slow growing lesion in the maxilla can mislead us to adenomatoid odontogenic tumor, however, the latter presents a unilocular radiolucency with an impacted canine or premolar.  Gibilisco suggests that bony spicules extend at right angles from the bone into the lesion with the surrounding bone intact (sun ray pattern), a feature that is pathognomonic of central hemangioma of the maxilla which serves to differentiate it from ameloblastoma, odontogenic myxoma, central giant cell granuloma, and others that have a honeycomb or soap bubble appearance. 
The age of the patient, site and recurrent nature of the swelling along with the radiographic features could mimic fibrous dysplasia, however, FNAC and other findings rule out the possibility. Although osteosarcoma has common radiographic appearance of our case, FNAC and contrast imaging strongly supports the lesion to be a central hemangioma. Similar to a central hemangioma, aneurysmal bone cyst also presents as a slow growing lesion, however, it is more common in the mandible showing aspiration positive with blood. Unlike central hemangioma, this lesion does not resorb the roots of the adjacent teeth.  Central hemangioma of bone may be clinically indistinguishable from other vascular conditions, such as shunts or aneurysms, but later conditions present with a painful growth of swelling. 
CT scan can differentiate the extensive speculate pattern of periosteal reaction in osteosarcoma, bone destruction, and strands of a fine, lace-like density in myxoma, partially cystic meshwork divided by coarse septae of ABC, and ground-glass appearance with ill-defined margins in fibrous dysplasia,  from the expansile lytic lesion wih trabeculations and thin bony spicules, suggesting a sunburst appearance of central hemangioma, which is expressed in this case. The relatively low signal on T1 weighted and low to intermediate signal on the T2 weighted images presented in this case are in contrast to the isointense T1 weighted and hyperintense T2 weighted images without flow voids and phleboliths that correspond to venous malformations. 
Hemangioma is a benign tumor, the greatest hazard being exsanguinating hemorrhage. , Treatment methods mentioned in the literature include: (1) noninvasive radiotherapy; (2) injection of sclerosing and embolizing agents;  and (3) surgical intervention by curettage and radical resection with immediate osseous reconstruction.  Other modes of treatment include steroid therapy, carbon dioxide and argon laser therapy, sclerosing agents  that provoke an inﬂammatory response with subsequent fibrosis and obliteration of vascular channels. The success of sclerosing agents is, however, restricted to superﬁcial soft tissues, and their value in treating intraosseous lesions is doubtful. 
| Conclusion|| |
Although conventional radiographic techniques reveal the classical radiographic features of central hemangioma, CT was primarily useful for illustrating the bony components of the tumor as well as delineating soft tissue extensions and bony defects which are not normally visible with plain film examination. Magnetic resonance imaging proved useful in demonstrating the vascular nature and flow voids representative of feeder vessels and delineating the true extent of the lesion, and, even more specifically, as low or high flow, suggesting a limited differential diagnosis. Central hemangioma should be treated without delay because trauma that disrupts the integrity of the affected jaw may result in lethal exsanguinations. Surgical curettage has nearly 20% recurrence rate, which can be reduced with complete removal of the lesion.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
We acknowledge the support of Lt. Gen. (Dr.) S. Murali Mohan, AVSM, M.D.S., FICD, Director, and Dr. V. Prabhakar M.D.S., Principal, Professor and HOD, Department of Conservative Dentistry, Sri Ramakrishna Dental College and Hospital, Coimbatore, Tamil Nadu, India, in preparation of this manuscript.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Shira RB, Guernsey LH. Central cavernous hemangioma of the mandible: Report of a case. J Oral Surg 1965;23:636-42.
Sadowsky D, Rosenberg RD, Kaufmen J, Levine BC, Friedman JM. Central haemangioma of the mandible. Literature review, case report, and discussion. Oral Surg Oral Med Oral Pathol 1981;52:471-7.
Hayward JR. Central cavernous haemangioma of the mandible: Report of four cases. J Oral Surg 1981;39:526-32.
White SC, Pharoah M. Benign tumors of the jaws. In: White SC, Pharoah M, editors. Oral radiology: Principles and interpretation. 4 th
ed. St Louis, Philadelphia, Sydney, Toronto: Mosby; 2000. p. 411-4.
Bunel K, Sindet-Pederson S. Central haemangioma of the mandible. Oral Surg Oral Med Oral Pathol 1993;75:565-70.
Marwah N, Agnihotri A, Dutta S. Central haemangioma: An overview and case report. Pediatr Dent 2006;28:460-6.
Beziat JL, Marcelino JP, Bascoulergue Y, Vitrey D. Central vascular malformation of the mandible: A case report. J Oral Maxillofac Surg 1997;55:415-9.
Wood NK, Goaz PW. Multilocular radiolucencies. In: Wood NK, Goaz PW, editors. Differential Diagnosis of Oral and Maxillofacial Lesions. 5 th
ed. St. Louis, Mo: The CV Mosby Company; 1997. p. 348-9.
Langland OE, Langlais RP, Mc David WD, Delbalso AM. Multilocular radiolucencies. In: Langland OE, Langlais RP, Mc David WD, Delbalso AM, editors. Panoramic Radiology. 2 nd
ed. Philadelphia, Pa: Lea & Febiger; 1989. p. 288-90.
Nagpal A, Suhas S, Ahsan A Pai KM, Rao NN. Central haemangioma: Variance in radiographic appearance. Dentomaxillofac Radiol 2005;34:120-5.
Kakimoto N, Tanimoto K, Nishiyama H, Murakami S, Furukawa S, Kreiborg S. CT and MR imaging features of oral and maxillofacial hemangioma and vascular malformation. Eur J Radiol 2005;55:108-12.
Gorlin RJ, Goldman HM. In: Thoma KH, Goldman HM, editors. Thoma's Oral Pathology. 6 th
ed. St. Louis, Mo: CV Mosby Company; 1971. p. 564-6.
Mody RN, Sathawane RS, Rai S. Central haemangioma. Review and a case report. Ann Dent 1995;54:22-4.
Kaneko R, Tohnai I, Ueda M, Negoro M, Yoshida J. Curative treatment of central haemangioma in the mandible by direct puncture and embolization with butyl-cyanoacrylate. Oral Oncol 2001;37:605-8.
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