The objective of this article is to elucidate on our experience with the first case reported in the literature of a synchronous presentation of pleomorphic adenoma (PA) in infratemporal fossa as well as the contralateral submandibular gland. PA most commonly arises from the major salivary glands but has also been reported to arise from minor salivary glands in unusual sites such as the nasal septum, main stem bronchus, trachea, lacrimal gland, external auditory canal, etc. However, it seldom involves the infratemporal fossa and extremely few cases are reported in the literature and coexistent contralateral synchronous PA is nowhere reported in literature. The infratemporal fossa tumour posed a diagnostic dilemma and therapeutic challenge because of its concealed location which we dealt successfully with endoscopic sublabial trans maxillary approach.
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