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Background/Objective Neuropathies may be rarely due to neurolymphomatosis. This can have several different pathological findings, such as epineural infiltration, endoneural perivascular inflammation, and axonal degeneration. We describe a patient with a polyneuropathy secondary to intravascular large B-cell lymphoma, with the sole pathology seen on biopsy being epineural intravascular infiltration.Methods Case study and literature review.Results A 61 year old male presented with a 2 week history of gradually progressive symmetric lower limb weakness resulting in loss of ambulation. Nerve conduction studies and electromyography revealed a severe, predominantly axonal non-length-dependent polyneuropathy, with motor fibres affected more than sensory fibres. A sural and muscle biopsy taken 2 weeks after onset of symptoms revealed intravascular large B-cell lymphoma. Interestingly, the nerve biopsy showed epineural vessel involvement, without evidence of nerve fibre pathology.Prednisone was started while awaiting the biopsy result, Subsequently, the patient‘s strength started to improve. The patient is being treated with chemotherapy.Conclusion This case highlights that in intravascular large B-cell lymphoma, histology may only demonstrate intravascular infiltration without nerve pathology. This suggests that the nerve pathology may be at a separate location, or yet to develop.