Abstract
IntroductionAmeloblastomas are rare lesions constituting 1% of all jaw tumors. Oral squamous cell carcinomas are common lesions; these constitute about 90% of all oral cancers. Concurrent tumors consisting of ameloblastoma and squamous cell carcinoma are extremely rare.Case presentationThis case report describes a 35-year-old African man who presented with a large mandibular tumor with an orocutaneous fistula that was found to be an ameloblastoma on histopathological examination, with concurrent squamous cell carcinoma histology within the fistula. This presentation was consistent with a Marjolin's ulcer within an ameloblastoma.ConclusionAmeloblastomas and Marjolin's ulcers require different management strategies. Careful histopathological examination of surgical specimens is key to patient outcome, as treatment of these patients depends on an accurate diagnosis.
Highlights
Histopathological examination of the tumor specimen revealed it to be an ameloblastoma with clear surgical margins, but it contained within it a squamous cell carcinoma limited to the orocutaneous fistula (Figures 3 and 4)
If the tumor occurs in an anatomical area with two apposing epithelialized surfaces such as the oral cavity and skin, it is feasible that tumor necrosis and ulceration into both epithelia might lead to the formation of a fistula
The fact that squamous cell carcinoma was found only along the orocutaneous fistula in this patient provides a strong basis for the hypothesis that chronic inflammation along the fistula over time led to malignant degeneration and to Marjolin’s ulcer (Figure 1)
Summary
Ameloblastoma is a benign but locally aggressive odontogenic tumor of the mandible and maxilla. Besides a history of having chewed khat for most of his adult life, the patient had no other identifiable risk factors for oral malignancy His physical examination revealed a wasted appearance with a large, ulcerated left- sided mandibular tumor that emitted a purulent, foul-smelling discharge (Figure 1). Histopathological examination of the tumor specimen revealed it to be an ameloblastoma with clear surgical margins, but it contained within it a squamous cell carcinoma limited to the orocutaneous fistula (Figures 3 and 4). Post-operatively, the patient did well and was offered left-sided mandibular reconstruction, but he was lost to follow-up after four months, until he re-presented to the hospital in healthy condition 13 months after surgery, with no evidence of either local recurrence or systemic tumor spread (Figure 5)
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