Caspr2 antibodies in a patient with prostate cancer: a cognitive deterioration with recurrent and paroxysmal gait ataxia and aphasia

Contactin-associated protein 2 (CASPR2) antibodies are originally associated with Morvan's syndrome and peripheral nerve hyper excitability. Our objective was to study retrospectively the clinical spectrum of CASPR2 antibody-positive patients in our hospital. This is a retrospective observational study. Patients treated at the Amrita Institute of Medical Sciences from May 2013 to April 2016, who were tested positive for CASPR2 antibodies, were included. A total of 1584 samples were tested in the neuroimmunology laboratory during the study period for voltage-gated potassium channel (VGKC) complex antibodies-leucine-rich glioma-inactivated protein 1 (LGI1) and CASPR2 antibodies. Thirty-four were positive for LGI1, 13 were positive for CASPR2, and 7 were for both (total 54-3.4% positivity). Of these 54 cases, 11 were treated in our hospital. Seven were positive for LGI1, three for CASPR2, and one for both. The patient who had both CASPR2 and LGI1 antibody positive had Morvan's syndrome. One patient with CASPR2 had neuromyotonia. The other patient was admitted with status epilepticus with a syndrome of parkinsonism and ataxia. The third patient had encephalopathy and myoclonus with a syndrome of parkinsonism and ataxia. Two of them underwent siddha treatment for other ailments prior to the onset of the disease for other ailments. Our short series shows the expanding spectrum of CASPR2 autoimmunity. Syndrome of parkinsonism and ataxia is an important manifestation of CASPR2 autoimmunity where we can offer a definitive treatment.

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Expanding spectrum of contactin-associated protein 2 (CASPR2) autoimmunity—syndrome of parkinsonism and ataxia

Kannoth

Nambiar

Gopinath

et al. 2017

Neurol Sci

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Morvan’s syndrome—is a pathogen behind the curtain?

et al. 2018

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Morvan's syndrome is a rare syndrome of likely autoimmune etiology characterized by peripheral nerve hyperexcitability, dysautonomia, insomnia, and fluctuating delirium with prominent hallucinations. Since its first mention in 1890, less than 100 cases have been described in literature. The largest existing review includes details of 29 cases. This case series describes 4 cases (M = 4) of Morvan's syndrome which presented between May and November 2017 to a single tertiary care referral teaching hospital in north India. All the four patients manifested behavioral abnormalities, sleep disturbances, hallucinations, autonomic dysfunction, and clinical signs of peripheral nerve hyperexcitability, mostly as myokymia. Two of the patients had Anti-CASPR2 (contactin-associated protein 2) antibodies. Three of them had electromyography features of peripheral nerve hyperexcitability and only one had elevated cerebrospinal fluid protein level. We hypothesize that Morvan's syndrome and other less characterized autoimmune encephalitis/peripheral nervous system syndromes may have infectious triggers. A possible viral trigger may result in generation of autoantibodies which result in the typical manifestations. We base these hypotheses on the finding of four cases of an orphan disease within a short period of time in a limited geographical distribution.

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Anti-contactin-associated protein-like 2 antibody-associated cerebellar ataxia: A case report and literature review

Wang

Qiu

et al. 2021

Journal of Neuroimmunology

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Caspr2 antibodies in a patient with prostate cancer: a cognitive deterioration with recurrent and paroxysmal gait ataxia and aphasia

Cited by 4 publications

References 6 publications

Expanding spectrum of contactin-associated protein 2 (CASPR2) autoimmunity—syndrome of parkinsonism and ataxia

Expanding spectrum of contactin-associated protein 2 (CASPR2) autoimmunity—syndrome of parkinsonism and ataxia

Morvan’s syndrome—is a pathogen behind the curtain?

Anti-contactin-associated protein-like 2 antibody-associated cerebellar ataxia: A case report and literature review

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