• Xintong Pang, Yanming Li, Zining Liu, Yafei Mao, Xinyuan Li, Lingling Gao, Yulan Geng, and Li Meng.
• Department of Laboratory Medicine, The First Hospital of Hebei Medical University, Shijiazhuang, China.
• Medicine (Baltimore). 2024 Jul 19; 103 (29): e38929e38929.

RationaleThe phenomenon of hypercoagulability has not been previously documented in individuals with Morvan's syndrome, especially in those associated with contactin-associated protein-like receptor 2 (CASPR2).Patient ConcernsA previously healthy 32-year-old Chinese male was admitted to the hospital with central and peripheral neurologic symptoms. The patient was tested positive for anti-CASPR2 antibodies, and also presented with an activated coagulation state on admission, characterized by a low activated partial thromboplastin time and a high platelet count. With gradual improvement of clinical symptoms, activated partial thromboplastin time, and platelet count returned to normal. Simultaneously, anti-CASPR2 antibody titers significantly decreased and eventually became undetectable.DiagnosesThe patient was diagnosed as Morvan's syndrome with positive anti-CASPAR2 antibodies accompanied with hypercoagulable state.InterventionsPlasmapheresis was administered to improve the symptoms combined with prednisolone acetate therapy.OutcomesThe patient experienced complete resolution of all symptoms during hospitalization and generally recovery after 2 months of discharge.LessonsEmphasis should be directed towards hypercoagulability in individuals diagnosed with Morvan's syndrome, particularly those presenting with positive anti-CASPR2 antibodies. Anticoagulant therapy may represent a novel therapeutic approach for individuals afflicted with Morvan's syndrome and exhibiting positivity for anti-CASPR2 antibodies.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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