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Lack of efficacy and off label use: case report A 44-year-old man exhibited lack of efficacy during treatment with immune-globulin for morvan syndrome. Additionally, he received off label therapy with rituximab for refractory morvan syndrome [dosages not stated; not all routes stated]. The man had a history of seropositive myasthenia gravis, recurrent thymoma, status-post thymectomy and chemotherapy. He presented with an acute-onset amnesia which followed a progressive course of fatigue accompanied by diffuse muscle cramps as well as twitching, limiting his ability to ambulate. Physical examination showed diffuse fasciculations and tachycardia. Antibody screening showed high serum and CSF titers of Contactin Associated Protein 2 (Caspr2) and Leucine-rich glioma-inactivated 1 (LGI1) antibodies. His findings were suggestive of morvan syndrome. The diffuse fasciculations and severe pain required treatment with gabapentin, morphine, and duloxetine. He failed to respond to two courses of IV immune-globulin and unspecified pulse steroid therapy. For refractory morvan syndrome, he started receiving off label treatment with rituximab. Following the first dose of rituximab, there was a dramatic improvement in the painful fasciculations and with the second dose, there was near complete resolution of signs and symptoms. At his last follow up, he remained in remission. Agha M, et al. Refractory morvan syndrome responding dramatically to rituximab. European Journal of Neurology 27 (Suppl. 1): 711 abstr. EPO1297, May 2020. Available 803498452 from: URL: https://onlinelibrary.wiley.com/toc/14681331/2020/27/9 [abstract]

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Reactions 29 Aug 2020 No. 1819