Conservative Management for Acanthomatous Ameloblastoma of Anterior Maxilla: A Rare Case Report
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Postgraduate Resident, Department of Oral and Maxillofacial Surgery, SGT University, Gurugram, Haryana, India.
First described by Broca in 1886, Ameloblastoma is one of the most frequently encountered epithelial odontogenic benign tumor. The literature reports Mandible as the utmost favourable area of this tumor with approximately 80% of the cases involving this area. Histologically, an ameloblastoma can have six variations i.e, follicular, plexiform, basal, granular, desmoplastic and/or acanthomatous form. According to the 2005 World Health Organization (WHO) classification the incidence of acanthomatous ameloblastoma is 7.06%. The acanthomatous type of ameloblastoma is deemed to be a rare and uncommon variation that mostly involves the mandible in 81% of the cases and maxilla in only 19% of the cases. In the maxilla, it is most commonly seen in the molar area, antral area and nasal floor with reported rate of occurrence being 47%, 33% and 9% respectively. The incidence in the canine are is only 9%. On the basis of previous studies, acanthomatous ameloblastoma is usually found in the geriatric population than in younger ones and is considered as an aggressive variant of ameloblastoma. Authors hereby present a case of acanthomatous ameloblastoma occurring in the anterior maxilla in a 36-year-old male patient presenting with painless swelling involving the left side of the anterior upper jaw managed with a conservative type of treatment. This type of presentation of the already rare acanthomatous ameloblastoma is extremely sporadic. Also, the occurrence of such a tumour in maxilla can significantly affect the progression and prognosis of such tumours because of the anatomical and histopathological differences in the maxilla and mandible and their densities, thereby further influencing the management and follow-up.
Adamantioma maxilla, Jaw, Neoplasms, Odontogenic, Tumour
A 36-year-old male reported to Department of Oral and Maxillofacial Surgery with the complaint of swelling on his left upper front jaw region evolving for 4 months, small at the beginning then gradually expanding in size. Patient gave no significant past dental or medical history. Also, no history of pain, trauma or discharge from swelling was elucidated.
On extraoral examination, a large swelling with diffused borders of the left superior labium was seen that obliterated the nasolabial fold (Table/Fig 1). It was approximately 2.5×2.5 cm in greatest dimension and extended from the left philtral ridge to the left nasolabial fold not crossing the midline anteroposteriorly and from the nasal floor to the vermilion on left side superiorinferiorly. The edges appeared indistinct; skin over the swelling appeared tense and glossy, with no change in colour. On palpation, the swelling was non tender, non compressible, and firm in consistency.
On intraoral examination single localised, round, sessile swelling approximately. 2×2 cm in size extending from 22 to 24 anteroposteriorly and mucobuccal fold to cervical margin of gingiva superioinferiorly was seen. On palpation, the swelling was non tender, non fluctuant, fixed and firm in consistency. There was slight rise in temperature and bleeding on probing. Vestibular obliteration with expansion of left buccal cortical plates in relation to 22 to 24 were noted (Table/Fig 2). Attrition in relation to 21 to 24 was also seen.
Radiological investigations, Orthopantomogram (OPG) and Cone Beam Computed Tomography were done. The OPG (Table/Fig 3) revealed a multilocular radiolucency with a honey comb pattern in the anterior region with displaced roots of 22 and 23. A cone beam computed tomography (Table/Fig 3) was done and it showed an osteolytic lesion that extended from distal side of 22 to mesial side of 23, displacing their roots. Expansion and break in continuity of buccal cortical plate and thinning of palatal cortical plate was noted. Based on the history, clinical and radiological features, a central giant cell granuloma was provisionally diagnosed. Ameloblastoma and odontogenic keratocyst were considered as differential diagnosis.
Incisional biopsy sample taken and sent for histopathological examination revealed periphery of the follicle lined by a layer of tall columnar ameloblast like cells with polarity of the nucleus away from the basement membrane. Loosely arranged polygonal or angular cells resembling stellate reticulum were seen in centre region. Ameloblastic epithelium surrounded large keratin filled cavity and many solid epithelial cell nests also showed squamous differentiation suggestive of acanthomatous ameloblastoma (Table/Fig 4)a-d.
After obtaining the consent from the patient, patient was taken for surgery under general anaesthesia. A conservative approach was followed. Crevicular incision extending from 11 to 24 was made. A full thickness mucoperiosteal flap was raised. Extraction of 22, 23 and 24 was done and lesion was enucleated along with curettage of the site (Table/Fig 5)a-d. Peripheral ostectomy was done to ensure complete removal. Reconstruction was done with iliac cancellous bone. Antibiotics, analgesics and anti-inflammatory drugs were given postoperatively. Histopathological examination of the excised specimen confirmed acanthomatous ameloblastoma. Wound healing was uneventful. Patient was reviewed 1 week and 1 month postoperatively and there after every 6 months for clinical and radiographic assessment. During follow-up, graft uptake was satisfactory and there were no signs of inflammation, infection or recurrence of the tumour clinically (Table/Fig 6). Radiographically, there were signs of graft maturation and new bone formation (Table/Fig 7).
Although ameloblastomas are among the commonest of odontogenic benign tumours, the incidence of occurrence of the acanthomataous variant of ameloblastoma in the anterior region of maxilla is extremely rare in human population. As per present knowledge, only a few cases of this type of ameloblastoma have been so far described in the literature. Ameloblastomas are aggressively treated owing to their locally destructive behaviour and propensity of reoccurrence, however, in present case it was managed conservatively and so far no recurrence is reported. However, further study of molecular mechanism, implication in clinical practice, and different treatment options have to be considered.
Date of Submission: Sep 09, 2021
Date of Peer Review: Dec 06, 2021
Date of Acceptance: Jan 20, 2022
Date of Publishing: May 01, 2022
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes
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