Introduction: Salivary gland tumors are complex to diagnose both on histopathology and cytology. Majority of these tumours arise in the parotid glands, followed by submandibular
glands, and rarely in minor salivary glands. Warthin’s tumor (WT), also known as papillary cystadenoma lymphomatosum, is characterized by a dense lymphoid stroma and a double
layer of oncocytic epithelium with papillary and cystic architecture. Only 6-10% cases have been reported in the extra-parotid locations i.e. cervical lymph nodes, submandibular gland and
larynx.

Case Report: Our patient presented with painless bilateral submandibular swellings for 1.5 months with history of chronic smoking of two bundles of biddis per day for 32 years.

Conclusion: Hereby, this case was unique in its presentation as it was located in an extra parotid region with bilateral origin, as these locations carry clinical significance in diagnosing
and ruling out conditions like lymphoma.