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Morvan Syndrome Converted from Isaacs' Syndrome after Thymectomy with Positivity for Both Anti-LGI1 and Anti-CASPR2 Antibodies
Authors:Daisuke Suzuki  Yoshihiro Suzuki  Daisuke Sato  Kenji Kikuchi  Naoki Kanauchi  Akiko Nishida  Yasuyuki Ohta
Affiliation:1.Department of Neurology, Nihonkai General Hospital, Japan; 2.Department of Respiratory Surgery, Nihonkai General Hospital, Japan; 3.Department of Pathology, Nihonkai General Hospital, Japan; 4.Division of Neurology and Clinical Neuroscience, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Japan
Abstract:
Anti-voltage-gated potassium channel complex antibodies-mediated disorder includes Isaacs'' syndrome, which is characterized by neuromyotonia, and Morvan syndrome, which is characterized by neuromyotonia, encephalopathy and autonomic dysfunction. We herein report a patient with Morvan syndrome that converted from Isaacs'' syndrome after thymectomy. The patient first presented with myospasm in all extremities and positivity for both anti-leucine-rich glioma inactivated 1 (LGI1) and anti-contactin-associated protein like 2 (CASPR2) antibodies and subsequently developed encephalopathy after thymectomy, which was successfully improved by immunotherapy. This is the first case of Morvan syndrome wherein thymectomy worsened Isaacs'' syndrome, suggesting that immunotherapy should be considered for Isaacs'' syndrome accompanied by positivity for both anti-LGI1 and anti-CASPR2 antibodies to prevent worsening to Morvan syndrome.
Keywords:CASPR2   Isaacs'' syndrome   LGI1   Morvan syndrome   thymectomy
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