Extra-nodal Non-Hodgkin’s Lymphomas (NHL) in the head and neck region are most commonly found in the Waldeyer’s lymphatic ring. However, Non-Hodgkin’s Lymphomas occurring in infra-temporal fossa are exceptionally rare. We report a case of a 38-year-old female patient, who presented with unilateral exophthalmos and facial paresthesia persisting over three months. Imaging and histological investigations revealed a primary Non-Hodgkin’s Lymphoma located in the infra-temporal region. The patient underwent chemotherapy with a favorable outcome observed over three years of follow-up. Through this case, we highlight the diagnostic challenges faced when dealing with a non-specific clinical and radiological presentation, as well as a biopsy site that is difficult to access. This case also underlines the need for a multidisciplinary approach involving ENT specialists, radiologists, and oncologists to address the diagnostic and therapeutic challenges associated with such rare presentation.
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