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論文
- タイトル
- タイトル（英）
- Late relapse of anti-N-methyl-d-aspartate receptor encephalitis with amusia and transiently reduced uptake in 123I-iomazenil single-photon emission computed tomography.
- 参照URL
- https://researchmap.jp/childbrain/published_papers/41612195
- 著者
- 著者（英）
- Naoki Yamada,Ichiro Kuki,Taeka Hattori,Naohiro Yamamoto,Shizuka Nagase,Megumi Nukui,Takeshi Inoue,Shin Okazaki,Hisashi Kawawaki,Asako Horino,Hiroshi Sakuma
- 担当区分
- 概要
- 概要（英）
- INTRODUCTION: Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis has a high relapse rate at approximately 10-20%. Most relapses occur within 2 years from onset, and 5 years after onset is rare. We report a case of anti-NMDAR encephalitis relapse with amusia 10 years after the initial encephalitis and discuss the usefulness of 123I-iomazenil single-photon emission computerized tomography (IMZ-SPECT) for its diagnosis. CASE: A 13-year-old left-handed girl presented with a depressed level of consciousness and focal to bilateral tonic-clonic seizures. Cerebrospinal fluid (CSF) analysis showed a mildly increased white blood cell count, elevated neopterin levels, and positive oligoclonal bands. Brain MRI was normal. IMZ-SPECT revealed reduced uptake in the right frontoparietal region. She received intravenous pulse methylprednisolone (IVMP) and high-dose intravenous immunoglobulin for autoimmune encephalitis; her symptoms resolved without neurological deficits. At 23 years old, she had mild right-sided numbness, dysarthria, amusia, and tonic-clonic seizures. Although the CSF analysis and brain MRI were normal, IMZ-SPECT revealed reduced uptake, indicating a relapse of encephalitis. IVMP administration resolved the symptoms. After discharge, the initial and relapse CSF analysis revealed anti-NMDAR antibodies. CONCLUSION: An anti-NMDAR encephalitis relapse 10 years after onset has never been reported. IMZ-SPECT may help in the diagnosis of anti-NMDAR encephalitis.
- 出版者・発行元
- 出版者・発行元（英）
- 誌名
- 誌名（英）
- Brain & development
- 巻
- 44
- 号
- 8
- 開始ページ
- 558
- 終了ページ
- 561
- 出版年月
- 2022年9月
- 査読の有無
- 招待の有無
- 掲載種別
- ISSN
- DOI URL
- https://doi.org/10.1016/j.braindev.2022.05.003
- 共同研究・競争的資金等の研究課題
研究者
佐久間 啓
(サクマ ヒロシ)