|
|
|
| Clinical analysis of anti-CASPR2 antibody associated autoimmune encephalitis#br# |
| JI Peng1 KOU Qixing1 SUN Gen1 QU Xueping1 JIANG Limin2▲ |
| 1.Department of Neurology, the Third People’s Hospital of Zhengzhou Tumor Hospital of Henan University, Henan Province, Zhengzhou 450000, China; 2.Department of Physical Examination, the First Affiliated Hospital of Henan University of Chinese Medicine, Henan Province, Zhengzhou 450000, China |
|
|
|
Abstract Objective To investigate the clinical features and treatment of anti-contactin associated protein-like 2(CASPR2) antibody associated autoimmune encephalitis. Methods Clinical data of five cases of anti-CASPR2 antibody associated autoimmune encephalitis diagnosed in the Third People’s Hospital of Zhengzhou and the First Affiliated Hospital of Henan University of Chinese Medicine from March 2017 to December 2020 were retrospectively analyzed. Results Among the five patients, there were three male patients, one patient had a history of fever before onset, two patients showed abnormal mental behavior, two patients showed epilepsy as the first symptom, and one patient showed memory loss. One patient had insomnia based on nerve pain, one patient had insomnia combined with cerebellar symptoms, and two patient had autonomic nerve dysfunction. Serum anti-CASPR2 antibody was positive in five patients, and cerebrospinal fluid anti-CASPR2 antibody was positive in one patient. All patients received immunotherapy, and the treatment was effective. Conclusion Anti-CASPR2 antibody associated autoimmune encephalitis may involve the central nervous system, the peripheral nervous system, the autonomic nervous system and other systems, leading to variable clinical syndrome, and immunotherapy is effective.
|
|
|
|
|
|
[1] van Sonderen A，Petit-Pedrol M，Dalmau J，et al. The value of LGI1，Caspr2 and voltage-gated potassium channel antibodies in encephalitis [J]. Nat Rev Neurol，2017，13（5）：290-301.
[2] 关鸿志，王佳伟.中国自身免疫性脑炎诊治专家共识[J].中华神经科杂志，2017，50（2）：91-98.
[3] Yuan J,Wang Y,Hu W，et al. The reliability and validity of a novel Chinese version simplified modified Rankin scale questionnaire (2011) [J]. BMC Neurol，2020，20（1）：127.
[4] Ellwardt E，Geber C，Lotz J，et al. Heterogeneous presentation of caspr2 antibody-associated peripheral neuropathy - A case series [J]. Eur J Pain，2020，24（7）：1411-1418.
[5] Zhang SJ，Xue YY，Yu H，et al. Morvan syndrome associated with LGI1 antibody：a case report[J]. BMC Neurol，2021，21（1）：185.
[6] Lai M，Huijbers MGM，Lancaster E，et al. Investigation of LGI1 as the antigen in limbic encephalitis previously attributed to potassium channels：a case series [J]. Lancet Neurol，2010，9（8）：776-785.
[7] van Sonderen A，Schreurs MWJ，Wirtz PW，et al. From VGKC to LGI1 and Caspr2 encephalitis：The evolution of a disease entity over time [J]. Autoimmunity Rev，2016，15（10）：970-974.
[8] Gillespie LE，Dave A，Goldstein A. A Tale of Two Brothers：Familial Voltage-Gated Potassium Channel Autoimmune Encephalitis [J]. Cureus，2020，12（6）：e8723.
[9] Michael S，Waters P，Irani SR. Stop testing for autoantibodies to the VGKC-complex：only request LGI1 and CASPR2 [J]. Pract Neurol，2020，20（5）：377-384.
[10] R?觟ssling R，Prüss H. Apheresis in Autoimmune Encephalitis and Autoimmune Dementia [J]. J Clin Med，2020， 9（9）：2683.
[11] Lancaster E. Autoimmune Encephalitis [J]. Decision-Making in Adult Neurology，2021：236-237.
[12] Boyko M，Au K，Casault C，et al. Systematic review of the clinical spectrum of CASPR2 antibody syndrome [J]. J Neurol，2020，267（4）：1137-1146.
[13] van Sonderen A，Ari?觡o H，Petit-Pedrol M，et al. The clinical spectrum of Caspr2 antibody-associated disease [J]. Neurology，2016，87（5）：521-528.
[14] 赵静，张元杏，刘慧勤，等. 抗接触蛋白相关蛋白-2抗体相关脑炎11例临床分析[J].中华神经科杂志，2019， 52（7）：549-554.
[15] Joubert B，Saint-Martin M，Noraz N，et al. Characterization of a Subtype of Autoimmune Encephalitis With Anti-Contactin-Associated Protein-like 2 Antibodies in the Cerebrospinal Fluid，Prominent Limbic Symptoms，and Seizures [J]. JAMA Neurol，2016，73（9）：1115-1124.
[16] Joubert B，Gobert F，Thomas L，et al. Autoimmune episodic ataxia in patients with anti-CASPR2 antibody-associated encephalitis [J]. Neurol Neuroimmunol Neuroinflamm，2017，4（4）：e371.
[17] Devine MF，Kothapalli N，Elkhooly M，et al. Paraneoplastic neurological syndromes：clinical presentations and management [J]. Ther Adv Neurol Disord，2021，14：1756 28642098532.
[18] Lee CH，Lin JJ，Lin KJ，et al. Caspr2 antibody limbic encephalitis is associated with Hashimoto thyroiditis and thymoma [J]. J Neurol Sci，2014，341（1/2）：36-40.
[19] Irani S R，Alexander S，Waters P，et al. Antibodies to Kv1 potassium channel-complex proteins leucine-rich, glioma inactivated 1 protein and contactin-associated protein-2 in limbic encephalitis, Morvan’s syndrome and acquired neuromyotonia [J]. Brain，2010，133（9）：2734-2748.
[20] Tüzün E，Kinay D，Hacohen Y，et al. Guillain-Barré-like syndrome associated with lung adenocarcinoma and CASPR2 antibodies [J]. Muscle Nerve，2013，48（5）：836-837.
[21] Vynogradova I，Savitski V，Heckmann JG. Hemichorea Associated with CASPR2 Antibody [J]. Tremor Other Hyperkinet Mov （N Y），2014，4：239.
[22] Gadoth A，Pittock SJ，Dubey D，et al. Expanded phenotypes and outcomes among 256 LGI1/CASPR2-IgG positive patients [J]. Ann Neurol，2017，82（1）：79-92．
[23] Gu Yixue，Zhong Min，He Liang，et al. Epidemiology of Antibody-Positive Autoimmune Encephalitis in Southwest China：A Multicenter Study [J]. Front Immunol，2019，10：2611.
[24] Bastiaansen AEM，van Sonderen A，Titulaer MJ. Autoimmune encephalitis with anti-leucine-rich glioma-inactivated 1 or anti-contactin-associated protein-like 2 antibodies (formerly called voltage-gated potassium channel-complex antibodies) [J]. Curr Opin Neurol，2017，30（3）：302-309.
[25] Dahm L，Ott C，Steiner J，et al. Seroprevalence of auto-antibodies against brain antigens in health and disease [J]. Ann Neurol，2014，76（1）：82-94.
[26] K?觟rtvelyessy P, Bauer J, Stoppel CM, et al. Complement-associated neuronal loss in a patient with CASPR2 antibody-associated encephalitis [J]. Neurol Neuroimmunol Neuroinflamm，2015，2（2）：e75. |
|
|
|
