Objective: To describe a unique case of hemichorea and oromandibular stereotypies as a possible side effect of immunotherapy for multiple myeloma.

Background: Multiple myeloma is a hematologic cancer of proliferating plasma cells often treated with immunomodulatory agents that target malignant plasma cells and induce cell death by a variety of mechanisms. While certain agents, such as lenalidomide, are qassociated with neurotoxicity, it usually involves the peripheral nervous system. Central nervous system toxicity, and in particular movement disorders, are rarely described in the natural history of multiple myeloma, or as a complication of treatment.

Method: This is a case report describing a single patient’s complication during treatment of multiple myeloma.

Results: A 76-year-old man developed severe hemichorea with ballistic features affecting the proximal left arm and leg as well as rhythmic, repetitive oromandibular and facial stereotypes resembling tardive dyskinesia in late 2024, 2 weeks after starting induction therapy for multiple myeloma with lenalidomide, daratumumab, bortezomib, and dexamethasone. He had not been exposed to any medication with dopamine receptor antagonist activity. Work up shortly after onset of the hyperkinetic movements included normal electrolytes, serum glucose, renal function, and blood counts. ALT and total bilirubin were mildly elevated but other liver function tests were normal. An MRI of the brain with contrast demonstrated midbrain and cerebellar atrophy, likely incidental, but no acute basal ganglia lesions. Cerebrospinal fluid (CSF) studies demonstrated a mild increase in CSF protein, without pleocytosis, and a negative infectious meningoencephalitis PCR panel. Serum antiphospholipid panel was negative. Stopping the chemotherapy and starting valbenazine eliminated the chorea and stereotypies within days. However, the abnormal movements resumed to a lesser degree about three weeks after reinitiating daratumumab, lenalidomide, and dexamethasone, without bortezomib, leading to a second discontinuation of the treatment.

Conclusion: Hemichorea and oromandibular stereotypies resembling tardive dyskinesia may be a rare side effect of multiple myeloma treatment with lenalidomide, daratumumab, and bortezomib, occurring as early as two weeks after initiating treatment, through an as yet undescribed pathologic mechanism.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/hemichorea-and-oromandibular-stereotypies-following-immunomodulatory-therapy-in-multiple-myeloma-a-case-report/