Hemangiomatous Ameloblastoma- A Case Report of a Very Rare Variant of Ameloblastoma

Abstract

A 35-year-old female patient presented with a chief complaint of painless swelling on the left side of the face since eight months to the Department of Oral Medicine and Radiology, Malla Reddy Dental College for Women, Hyderabad. Telangana, India. Her history revealed that the swelling gradually grew to the present size. Her medical, dental, habitual and family history was not significant. On extra oral examination, inspection showed a swelling measuring approximately 4x3 cm in size, oval in shape, having well defined margins. The colour of the lesion was similar to the normal adjacent skin. The swelling extended superiorly to the line extending from the tragus of the ear to the corner of the mouth. Inferiorly to lower border of the mandible, posteriorly up to the ramus of the mandible and anteriorly up to the corner of the mouth [Table/ Fig-1]. No visible pus discharge was seen. On palpation, the inspectory findings were confirmed and the lesion was bony hard with central region showing variable consistency of hard and soft areas. The lesion was non tender. On intra oral examination, firm, smooth, non-fluctuant swelling was seen extending from 31 to the anterior border of the ramus. The mandibular left first molar and second premolar have been extracted 11 months back. First premolar was carious.Panaromic radiograph revealed an well defined mixed radioopaquelucent multilocular lesion extending from lower left central incisor to the ascending border of ramus [Table/ Fig-2]. Impacted second molar was seen at the inferior border of the mandible. Few septae were arranged in tennis raquet appearance. Root resorption was noticed in relation to 34. Over retained 85 was also seen. Radiological differential diagnosis of central hemangioma, ameloblastoma, odontogenic myxoma was considered.A provisional diagnosis of ameloblastoma and clinical differential diagnosis of dentigerous cyst and odontogenic keratocyst was made. After obtaining patient's consent, enucleation was done and multiple bits of the soft tissue specimen of approximately 4cm X 2cm X 1cm were sent to the Department of Oral Pathology.On microscopic examination, the tissue section revealed plexiform ameloblastoma with a prominent vascular component. It showed anastomosing cords and sheets of odontogenic epithelium in a loosely arranged vascular connective tissue stroma. The epithelium was surrounded by cubodial ameloblast like cells with central stellate reticulum like cells. The vascular component consisted of blood filled spaces of varying sizes lined by endothelial cells. At focal areas vascular spaces were large and compressing the odontogenic strands . A diagnosis of Hemangiomatous Amelobalstoma was made.Partial hemimandibulectomy was planned followed by immediate reconstruction with a nonvascularised illiac graft. But the patient was not cooperative and reluctant for surgical treatment and hence the procedure could not be carried out. The patient was recalled for follow up visits but did not return to the department. DisCussionAmeloblastoma is one of t...

Hemangiomatous ameloblastoma: Case report with a brief review

“…HA can occur at any age but is most commonly seen in the third and fourth decades of life with mandibular posterior region as the most common site which accords with the present case which had occurred in a 35-year-old patient in the right mandibular region. Previous reports showed male preponderance,[ 3 4 11 12 13 14 15 ] but in this case was a female. In the present case, the radiographic appearance showed mixed radio-opaque– radiolucent lesion which is consistent with other cases reported by Kasangari et al .,[ 3 ] Harshavardhan et al .,[ 13 ] and Rajmohan et al .…”

“…Previous reports showed male preponderance,[ 3 4 11 12 13 14 15 ] but in this case was a female. In the present case, the radiographic appearance showed mixed radio-opaque– radiolucent lesion which is consistent with other cases reported by Kasangari et al .,[ 3 ] Harshavardhan et al .,[ 13 ] and Rajmohan et al . [ 15 ]…”

“…Hemangiomatous ameloblastoma term refers to any ameloblastoma which has many spaces filled with blood or large endothelial-lined capillaries in its stroma. [ 3 ] The first case of HA was described in 1932 by Kuhn as a combination of hemangioma and adamantinoma. [ 5 ] Later in 1950, Aisenberg reported a similar lesion by the term admantinohemangioma.…”

“…It is also suggested that excessive stimulation of angiogenesis during tumor development by inductive influences such as those that occur during odontogenesis or by other factors may result in the overgrowth of vascular elements in the odontogenic ectomesenchyme or in adjacent connective tissue. [ 12 ] Kasangari et al .,[ 3 ] Gargi et al . [ 4 ] and Rajmohan et al .…”

“…[ 2 ] A variable number of histopathological patterns of ameloblastoma exist and the common being follicular and plexiform patterns. [ 3 ] In the literature, a rare variant called hemangiomatous ameloblastoma (HA) was originally described as an ameloblastoma in which the tumor stroma contained spaces filled with blood or large endothelial-lined capillaries. [ 4 ] Only ten cases have been documented earlier in the literature.…”