Objective: To describe a case of delayed onset Orthostatic Myoclonus in a patient after withdrawal of checkpoint inhibitor immunotherapy.

Background: Movement disorders have been described in oncologic disease due to various etiologies – structural or compressive, paraneoplastic, and immune checkpoint inhibitor related adverse events.

Method: Myoclonus and ataxia have been described as possible neurological complications in patients treated with immune checkpoint inhibitors, but orthostatic myoclonus has not yet been reported. Here we describe the clinical history, workup, and phenomenology of a delayed onset orthostatic myoclonus after withdrawal of checkpoint inhibitor immunotherapy.

Results: A 71-year-old male with history of melanoma metastatic to the CNS with prior treatment with nivolumab/ipilimumab presented with jerking movements of the legs upon standing. Seven years prior to symptom onset, the patient had been diagnosed with melanoma metastatic to the left axillary lymph nodes, frontoparietal skull and adjacent dura. He was treated with pembrolizumab, then after progression of disease was switched to nivolumab/ipilimumab. This treatment was discontinued 5 months prior to the patient’s symptoms due to transaminitis. After onset of symptoms extensive workup was performed: two separate MRIs with and without contrast of the entire neuroaxis were unchanged from prior, two separate serum and CSF paraneoplastic/autoimmune encephalopathy panels were pan negative, and CT Chest Abdomen Pelvis with contrast obtained near onset of symptoms was without reoccurrence of disease. He was treated with high dose steroids for a presumed immune related adverse event with rapid improvement of symptoms. Given the temporal association of symptoms to withdrawal of immunotherapy, the negative paraneoplastic and structural workups, and the rapid steroid responsiveness of these symptoms – this case may represent a delayed neuro immune related adverse event.

Conclusion: Immune checkpoint inhibitors can cause various neurological adverse events. Recognition of movement disorders as a possible delayed neurologic side effect of immune checkpoint inhibitor may inform workup and treatment options.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/ipilimumab-nivolumab-related-orthostatic-myoclonus-in-a-patient-with-metastatic-melanoma/