Xiaohong Qin1,2,3, Jieying Li4,5, Yuanyuan Luo4,5, Yunsen He6, Xiaoqiang Xiao4,5, Arui Tan4,5, Jun Xiao7,8,9. 1. Sichuan Provincial Center for Mental Health, Sichuan Academy of Medical Science and Sichuan Provincial People's Hospital, Chengdu, 610072, China. 1005476612@qq.com. 2. Key Laboratory of Psychosomatic Medicine, Chinese Academy of Medical Sciences, Chengdu, 610072, China. 1005476612@qq.com. 3. Sichuan Provincial Center for Mental Health, Sichuan Academy of Medical Science and Sichuan Provincial People's Hospital, No.33, 2Nd Road, Furong AvenueWenjiang District, Chengdu, 611731, Sichuan Province, China. 1005476612@qq.com. 4. Sichuan Provincial Center for Mental Health, Sichuan Academy of Medical Science and Sichuan Provincial People's Hospital, Chengdu, 610072, China. 5. Key Laboratory of Psychosomatic Medicine, Chinese Academy of Medical Sciences, Chengdu, 610072, China. 6. Department of Neurosurgery, Sichuan Provincial People's Hospital, University of Electronic Science and Technology of China, Chengdu, 610072, China. 7. Sichuan Provincial Center for Mental Health, Sichuan Academy of Medical Science and Sichuan Provincial People's Hospital, Chengdu, 610072, China. 1458935803@qq.com. 8. Key Laboratory of Psychosomatic Medicine, Chinese Academy of Medical Sciences, Chengdu, 610072, China. 1458935803@qq.com. 9. Sichuan Provincial Center for Mental Health, Sichuan Academy of Medical Science and Sichuan Provincial People's Hospital, No.33, 2Nd Road, Furong AvenueWenjiang District, Chengdu, 611731, Sichuan Province, China. 1458935803@qq.com.
Abstract
OBJECTIVE: Anti-contactin-associated protein-like 2 (CASPR2) antibody encephalitis is a rare autoimmune encephalitis (AE) that often presents with epilepsy, cognitive dysfunction, peripheral neuropathy, autonomic nerve damage, and ataxia. Parkinsonism is often observed in neurodegenerative diseases but progresses slowly, and rapidly progressive parkinsonism is rare. Given that it is a curable parkinsonism, identifying and providing early immunotherapy is crucial. METHODS: We reported a patient initially presenting with anxiety and depression, whose symptoms were relieved following mood regulation treatment. After discontinuation of the mood-regulating drugs, mood disorders recurred, accompanied by parkinsonism. The onset of parkinsonism was subacute (< 3-month disease course), and progression was rapid. After immunotherapy, all symptoms disappeared completely. We reviewed all relevant literature on anti-CASPR2 antibody encephalitis with parkinsonism. RESULTS: Our literature review revealed three cases (including our patient): two male and one female, ranging in age from 48 to 72 years. All patients had parkinsonism, generalized tonic-clonic seizures, and hyponatremia. Three patients had anti-CASPR2 antibody positivity in the serum, and one patient had anti-CASPR2 antibody positivity in the CSF. All three patients were treated with anti-epileptic drugs and intravenous steroid pulse therapy, followed by oral steroid therapy, symptoms improved. CONCLUSION: Parkinsonism can be easily misdiagnosed as a neurodegenerative disease, especially during the early stages. In patients with parkinsonism, treatable diseases should be considered in addition to neurodegenerative diseases. In clinical practice, anti-CASPR2 antibody encephalitis should be considered if rapidly progressing parkinsonism is encountered after ruling out common etiologies.
OBJECTIVE: Anti-contactin-associated protein-like 2 (CASPR2) antibody encephalitis is a rare autoimmune encephalitis (AE) that often presents with epilepsy, cognitive dysfunction, peripheral neuropathy, autonomic nerve damage, and ataxia. Parkinsonism is often observed in neurodegenerative diseases but progresses slowly, and rapidly progressive parkinsonism is rare. Given that it is a curable parkinsonism, identifying and providing early immunotherapy is crucial. METHODS: We reported a patient initially presenting with anxiety and depression, whose symptoms were relieved following mood regulation treatment. After discontinuation of the mood-regulating drugs, mood disorders recurred, accompanied by parkinsonism. The onset of parkinsonism was subacute (< 3-month disease course), and progression was rapid. After immunotherapy, all symptoms disappeared completely. We reviewed all relevant literature on anti-CASPR2 antibody encephalitis with parkinsonism. RESULTS: Our literature review revealed three cases (including our patient): two male and one female, ranging in age from 48 to 72 years. All patients had parkinsonism, generalized tonic-clonic seizures, and hyponatremia. Three patients had anti-CASPR2 antibody positivity in the serum, and one patient had anti-CASPR2 antibody positivity in the CSF. All three patients were treated with anti-epileptic drugs and intravenous steroid pulse therapy, followed by oral steroid therapy, symptoms improved. CONCLUSION: Parkinsonism can be easily misdiagnosed as a neurodegenerative disease, especially during the early stages. In patients with parkinsonism, treatable diseases should be considered in addition to neurodegenerative diseases. In clinical practice, anti-CASPR2 antibody encephalitis should be considered if rapidly progressing parkinsonism is encountered after ruling out common etiologies.
Authors: Francesc Graus; Maarten J Titulaer; Ramani Balu; Susanne Benseler; Christian G Bien; Tania Cellucci; Irene Cortese; Russell C Dale; Jeffrey M Gelfand; Michael Geschwind; Carol A Glaser; Jerome Honnorat; Romana Höftberger; Takahiro Iizuka; Sarosh R Irani; Eric Lancaster; Frank Leypoldt; Harald Prüss; Alexander Rae-Grant; Markus Reindl; Myrna R Rosenfeld; Kevin Rostásy; Albert Saiz; Arun Venkatesan; Angela Vincent; Klaus-Peter Wandinger; Patrick Waters; Josep Dalmau Journal: Lancet Neurol Date: 2016-02-20 Impact factor: 44.182