Autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report

Autoimmune basal ganglia encephalitis causes neurological symptoms such as parkinsonism associated with basal ganglia lesions. Here, we report a case of autoimmune basal ganglia encephalitis without retinal lesions or malignancy harboring anti-recoverin antibodies. The patient was a 67-year-old Japa...

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Autores principales: Yuki Kitazaki, Norimichi Shirafuji, Naoko Takaku, Tomohisa Yamaguchi, Soichi Enomoto, Masamichi Ikawa, Osamu Yamamura, Yasunari Nakamoto, Tadanori Hamano
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Publicado: Elsevier 2021
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spelling oai:doaj.org-article:433a536754e040c68ca4ac211ccd7fc82021-11-26T04:35:31ZAutoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report2405-650210.1016/j.ensci.2021.100382https://doaj.org/article/433a536754e040c68ca4ac211ccd7fc82021-12-01T00:00:00Zhttp://www.sciencedirect.com/science/article/pii/S2405650221000757https://doaj.org/toc/2405-6502Autoimmune basal ganglia encephalitis causes neurological symptoms such as parkinsonism associated with basal ganglia lesions. Here, we report a case of autoimmune basal ganglia encephalitis without retinal lesions or malignancy harboring anti-recoverin antibodies. The patient was a 67-year-old Japanese woman who developed anorexia, parkinsonism, and disturbance of consciousness 7 days before admission. Brain magnetic resonance imaging showed hyperintense bilateral basal ganglia lesions on fluid-attenuated inversion recovery images. 18F-fluorodeoxyglucose-positron emission tomography showed no malignancy in the trunk, and dopamine transporter single-photon emission computed tomography with dopamine transporters revealed reduced radiotracer uptake in the basal ganglia. Further, anti-recoverin IgG antibodies were detected in serum immunoblot. Based on the clinical and imaging findings, the patient was diagnosed with autoimmune basal ganglia encephalitis with anti-recoverin antibodies and administered high-dose immunoglobulins (HD-IVIG), which led to an improvement in clinical symptoms. Anti-recoverin antibodies are paraneoplastic antibodies that explicitly bind to Ca2+-binding proteins in the retina and cause retinopathy. This pathological sequence is defined as cancer-associated retinopathy (CAR). However, in our case, autoimmune basal ganglia encephalitis developed without CAR syndrome or malignancy. Clinicians should be aware of the possibility of autoimmune basal ganglia encephalitis showing anti-recoverin antibodies but no CAR syndrome or malignancy, which should be treated with HD-IVIG therapy.Yuki KitazakiNorimichi ShirafujiNaoko TakakuTomohisa YamaguchiSoichi EnomotoMasamichi IkawaOsamu YamamuraYasunari NakamotoTadanori HamanoElsevierarticleAutoimmune basal ganglia encephalitisParkinsonismAutoimmune encephalitisHigh-dose immunoglobulinAnti-recoverin antibodiesNeurology. Diseases of the nervous systemRC346-429ENeNeurologicalSci, Vol 25, Iss , Pp 100382- (2021)
institution DOAJ
collection DOAJ
language EN
topic Autoimmune basal ganglia encephalitis
Parkinsonism
Autoimmune encephalitis
High-dose immunoglobulin
Anti-recoverin antibodies
Neurology. Diseases of the nervous system
RC346-429
spellingShingle Autoimmune basal ganglia encephalitis
Parkinsonism
Autoimmune encephalitis
High-dose immunoglobulin
Anti-recoverin antibodies
Neurology. Diseases of the nervous system
RC346-429
Yuki Kitazaki
Norimichi Shirafuji
Naoko Takaku
Tomohisa Yamaguchi
Soichi Enomoto
Masamichi Ikawa
Osamu Yamamura
Yasunari Nakamoto
Tadanori Hamano
Autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report
description Autoimmune basal ganglia encephalitis causes neurological symptoms such as parkinsonism associated with basal ganglia lesions. Here, we report a case of autoimmune basal ganglia encephalitis without retinal lesions or malignancy harboring anti-recoverin antibodies. The patient was a 67-year-old Japanese woman who developed anorexia, parkinsonism, and disturbance of consciousness 7 days before admission. Brain magnetic resonance imaging showed hyperintense bilateral basal ganglia lesions on fluid-attenuated inversion recovery images. 18F-fluorodeoxyglucose-positron emission tomography showed no malignancy in the trunk, and dopamine transporter single-photon emission computed tomography with dopamine transporters revealed reduced radiotracer uptake in the basal ganglia. Further, anti-recoverin IgG antibodies were detected in serum immunoblot. Based on the clinical and imaging findings, the patient was diagnosed with autoimmune basal ganglia encephalitis with anti-recoverin antibodies and administered high-dose immunoglobulins (HD-IVIG), which led to an improvement in clinical symptoms. Anti-recoverin antibodies are paraneoplastic antibodies that explicitly bind to Ca2+-binding proteins in the retina and cause retinopathy. This pathological sequence is defined as cancer-associated retinopathy (CAR). However, in our case, autoimmune basal ganglia encephalitis developed without CAR syndrome or malignancy. Clinicians should be aware of the possibility of autoimmune basal ganglia encephalitis showing anti-recoverin antibodies but no CAR syndrome or malignancy, which should be treated with HD-IVIG therapy.
format article
author Yuki Kitazaki
Norimichi Shirafuji
Naoko Takaku
Tomohisa Yamaguchi
Soichi Enomoto
Masamichi Ikawa
Osamu Yamamura
Yasunari Nakamoto
Tadanori Hamano
author_facet Yuki Kitazaki
Norimichi Shirafuji
Naoko Takaku
Tomohisa Yamaguchi
Soichi Enomoto
Masamichi Ikawa
Osamu Yamamura
Yasunari Nakamoto
Tadanori Hamano
author_sort Yuki Kitazaki
title Autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report
title_short Autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report
title_full Autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report
title_fullStr Autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report
title_full_unstemmed Autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: A case report
title_sort autoimmune basal ganglia encephalitis associated with anti-recoverin antibodies: a case report
publisher Elsevier
publishDate 2021
url https://doaj.org/article/433a536754e040c68ca4ac211ccd7fc8
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