SummaryBackground: Lhermitte's sign (LS) is characterized by electric shock-like sensations, spreading along the spine in a cervico-caudal direction and into the arms and legs. It is felt upon flexion of the neck and reflects a myelopathy resulting from damage to sensory axons at the dorsal columns of the cervical spinal cord. In patients with cancer, LS can occur due to direct tumour involvement of the spine or in relation to radiotherapy. Chemotherapy-related LS has only rarely been reported. Case Report: We present the case of a 44-year-old patient treated with platinum- and etoposide-based chemotherapy for metastatic seminoma, who subsequently developed severe disabling LS. The severity and duration of LS in this case exceeds prior reports. We review the literature on chemotherapy-related LS and discuss the practice implications. Conclusion: Neuropathy and LS related to cisplatin-based chemotherapy can result in significant adverse effects. Raised awareness of this complication could aid sub-phenotyping of the population most at risk and assist strategies to avert discomfort and disability post chemotherapy.