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Febrile infection-related epilepsy syndrome with claustrum lesion: an underdiagnosed inflammatory encephalopathy.

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Title: Febrile infection-related epilepsy syndrome with claustrum lesion: an underdiagnosed inflammatory encephalopathy.
Authors: Bai, Lin (AUTHOR), Di, Weiying (AUTHOR), Xu, Zucai (AUTHOR), Liu, Bin (AUTHOR), Lin, Nan (AUTHOR), Fan, Siyuan (AUTHOR), Ren, Haitao (AUTHOR), Lu, Qiang (AUTHOR), Wang, Jiawei (AUTHOR), Guan, Hongzhi (AUTHOR)
Source: Neurological Sciences. Jul2024, Vol. 45 Issue 7, p3411-3419. 9p.
Subjects: Epilepsy, Brain diseases, Viral encephalitis, Symptoms, Clinical indications, Signal detection
Abstract: Objective: To summarize the clinical characteristics and prognosis of febrile infection-related epilepsy syndrome with claustrum lesions (FIRES-C). Method: Clinical data of FIRES-C patients were collected retrospectively. The study reviewed and analyzed their clinical manifestations, treatment strategies, and prognosis. Result: Twenty patients were enrolled, including 13 females and 7 males, with a median onset age of 20.5 years. All patients developed seizures after fever, with a median interval of 5 days. Brain MRI showed symmetric lesions in the claustrum in all patients. The median interval from seizure onset to abnormal MRI signals detection was 12.5 days. All patients had negative results for comprehensive tests of neurotropic viruses and antineuronal autoantibodies. Seventy percent of cases had been previously empirically diagnosed with autoimmune encephalitis or viral encephalitis before. All patients received anti-seizure medicine. Eleven patients (55%) received antiviral therapy. All patients received immunotherapy, including glucocorticoids (100%), intravenous immunoglobulin (IVIg) (65%), plasma exchange (PLEX) (10%), tocilizumab (10%), rituximab (5%), and cyclophosphamide (5%). Sixty percent of patients received long-term immunotherapy (≥ 3 months). The median follow-up was 11.5 months;60% of patients were diagnosed with refractory epilepsy. Conclusion: Bilateral claustrum lesion on MRI is a distinctive neuroimage feature for FIRES, which may serve as an indication for the initial clinical assessments. FIRES-C should be classified as a type of inflammatory encephalopathy characterized by a monophasic nature. Some FIRES-C patients respond to immunotherapy and antiseizure treatments but most experience refractory epilepsy as a long-term outcome. [ABSTRACT FROM AUTHOR]
Copyright of Neurological Sciences is the property of Springer Nature and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)
Database: Psychology and Behavioral Sciences Collection
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  Data: Febrile infection-related epilepsy syndrome with claustrum lesion: an underdiagnosed inflammatory encephalopathy.
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  Data: <searchLink fieldCode="AR" term="%22Bai%2C+Lin%22">Bai, Lin</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Di%2C+Weiying%22">Di, Weiying</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Xu%2C+Zucai%22">Xu, Zucai</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Liu%2C+Bin%22">Liu, Bin</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Lin%2C+Nan%22">Lin, Nan</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Fan%2C+Siyuan%22">Fan, Siyuan</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Ren%2C+Haitao%22">Ren, Haitao</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Lu%2C+Qiang%22">Lu, Qiang</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Wang%2C+Jiawei%22">Wang, Jiawei</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Guan%2C+Hongzhi%22">Guan, Hongzhi</searchLink> (AUTHOR)
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  Data: <searchLink fieldCode="JN" term="%22Neurological+Sciences%22">Neurological Sciences</searchLink>. Jul2024, Vol. 45 Issue 7, p3411-3419. 9p.
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  Data: <searchLink fieldCode="DE" term="%22Epilepsy%22">Epilepsy</searchLink><br /><searchLink fieldCode="DE" term="%22Brain+diseases%22">Brain diseases</searchLink><br /><searchLink fieldCode="DE" term="%22Viral+encephalitis%22">Viral encephalitis</searchLink><br /><searchLink fieldCode="DE" term="%22Symptoms%22">Symptoms</searchLink><br /><searchLink fieldCode="DE" term="%22Clinical+indications%22">Clinical indications</searchLink><br /><searchLink fieldCode="DE" term="%22Signal+detection%22">Signal detection</searchLink>
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  Label: Abstract
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  Data: Objective: To summarize the clinical characteristics and prognosis of febrile infection-related epilepsy syndrome with claustrum lesions (FIRES-C). Method: Clinical data of FIRES-C patients were collected retrospectively. The study reviewed and analyzed their clinical manifestations, treatment strategies, and prognosis. Result: Twenty patients were enrolled, including 13 females and 7 males, with a median onset age of 20.5 years. All patients developed seizures after fever, with a median interval of 5 days. Brain MRI showed symmetric lesions in the claustrum in all patients. The median interval from seizure onset to abnormal MRI signals detection was 12.5 days. All patients had negative results for comprehensive tests of neurotropic viruses and antineuronal autoantibodies. Seventy percent of cases had been previously empirically diagnosed with autoimmune encephalitis or viral encephalitis before. All patients received anti-seizure medicine. Eleven patients (55%) received antiviral therapy. All patients received immunotherapy, including glucocorticoids (100%), intravenous immunoglobulin (IVIg) (65%), plasma exchange (PLEX) (10%), tocilizumab (10%), rituximab (5%), and cyclophosphamide (5%). Sixty percent of patients received long-term immunotherapy (≥ 3 months). The median follow-up was 11.5 months;60% of patients were diagnosed with refractory epilepsy. Conclusion: Bilateral claustrum lesion on MRI is a distinctive neuroimage feature for FIRES, which may serve as an indication for the initial clinical assessments. FIRES-C should be classified as a type of inflammatory encephalopathy characterized by a monophasic nature. Some FIRES-C patients respond to immunotherapy and antiseizure treatments but most experience refractory epilepsy as a long-term outcome. [ABSTRACT FROM AUTHOR]
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  Data: <i>Copyright of Neurological Sciences is the property of Springer Nature and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract.</i> (Copyright applies to all Abstracts.)
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              Text: Jul2024
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