Maxillary Ameloblastoma with Orbital Involvement: An Institutional Experience and Literature Review

Abstract

Purpose To describe 8 patients with orbital involvement by ameloblastoma and to review the literature on this topic. Methods The electronic medical records and pathology databases of the Hospital of the University of Pennsylvania were searched to identify all patients with histopathologically confirmed ameloblastoma diagnosed between 1990 and 2015. PubMed database was searched for all well-documented cases of maxillary ameloblastoma and ameloblastic carcinoma exameloblastoma with orbital involvement published in the English literature. The information collected on the compiled 23 patients included age, sex, clinical presentation, imaging findings, management, tumor histopathologic features, and follow up. Results Review of medical records identified 8 patients with orbital involvement by ameloblastoma. Literature search yielded 15 patients with well-documented orbital involvement by ameloblastoma. Most tumors occurred in men (19 of 23, M:F = 4–5:1) with an average age of 56 years. The overall rates of recurrence, visual compromise, death, and confirmed disease-related mortality were 70% (16/23), 26% (6/23), 39% (9/23), and 22% (5/23), respectively. The initial surgical approach correlated with prognosis. The rates of recurrence, orbital exenteration, and mortality in the cohort managed with conservative surgery or partial maxillectomy were 57% (8/14), 29% (4/14), and 50% (7/14), respectively. In contrast, the patients initially managed with a radical resection had substantially lower frequencies of tumor recurrence (2/7, 29%), exenteration (1/7, 14%), and death (1/7, 14%). Malignant transformation to ameloblastic carcinoma occurred in the setting of recurrent disease in 3 patients and in 1 patient with prolonged duration of symptoms, suggestive of a long-standing tumor. Conclusions Maxillary ameloblastoma can rarely involve the orbit, leading to significant ocular morbidity and occasional mortality. Prompt radical resection of the tumor has the potential to decrease the likelihood of recurrence and visual compromise, and can improve survival.

Ameloblastomas vs recurrent ameloblastomas: a systematic review

“…This is similar to the average age of occurrence of ameloblastomas and implies that recurrences occur within a few years of treatment. Arotiba et al [17] and Milman et al [12] reported highest recurrence in the third and fourth decade. Recurrent cases showed male predilection with male female ratio similar to that of ameloblastomas.…”

“…A total of 1206 cases of ameloblastoma were identified in 10 publications. Summary of studies included in the review regarding ameloblastoma in general and the recurrence rate is shown in Table I [4][5][6][7][8][9][10][11][12][13][14].…”

Management of Recurrence of Ameloblastoma and Odontogenic Keratocyst: A Cross-Sectional Study

“…Although ODOVTOS-International Journal of Dental Sciences radical surgery, including segmental or marginal resection of the jaw, is generally recommended, some authors have advocated more conservative methods as the first line treatment modality in some cases (3,12,23). Initial surgical approach correlated with risk of recurrence, local disease aggressiveness, and mortality (30), and enucleation exhibited most high recurrence rate in our study. Any recurrence will increase the degree of tissue damage and the need for further surgery.…”

A rare case of maxillary ameloblastoma with lung metastasis: a case report

“…Ameloblastomas are low-grade neoplasms of odontogenic epithelium that account for about 1% of all oral tumours and about 10% of odontogenic tumours [1]. They commonly occur in the mandible and involvement of the maxilla is reported to be about 20% [2,3]. Although they are considered to be benign tumours with a slow growth rate, they are locally aggressive with a high tendency for local recurrence if not removed completely [1,4].…”

“…Although they are considered to be benign tumours with a slow growth rate, they are locally aggressive with a high tendency for local recurrence if not removed completely [1,4]. Rarely, these tumours have the potential of distance metastasis [3], whereby this phenomenon is associated with a prolonged tumour duration and multiple recurrences following surgical interventions [2]. When histologically benign-appearing ameloblastoma does metastasize, it is referred to as metastatic ameloblastoma [5].…”