A case of Vogt-Koyanagi-Harada disease associated with non-herpetic acute limbic encephalitis with autoantibodies against glutamate receptor .EPSILON.2 in the cerebrospinal fluid

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  • Masuda Teruaki
    Department of Internal Medicine III, Oita University, Faculty of Medicine
  • Kimura Noriyuki
    Department of Internal Medicine III, Oita University, Faculty of Medicine
  • Ishibashi Masato
    Department of Internal Medicine III, Oita University, Faculty of Medicine
  • Ito Machiko
    Department of Ophthalmology, Oita University, Faculty of Medicine
  • Takahashi Yukitoshi
    Department of Pediatrics, National Epilepsy Center, Institute of Epilepsy and Neurological Disorders
  • Kumamoto Toshihide
    Department of Internal Medicine III, Oita University, Faculty of Medicine

Bibliographic Information

Other Title
  • 髄液抗グルタミン酸受容体ε２抗体陽性の非ヘルペス性急性辺縁系脳炎をともなったＶｏｇｔ－小柳－原田病の１例
  • 症例報告 髄液抗グルタミン酸受容体ε2抗体陽性の非ヘルペス性急性辺縁系脳炎をともなったVogt-小柳-原田病の1例
  • ショウレイ ホウコク ズイエキ コウグルタミンサン ジュヨウタイ イプシロン 2 コウタイ ヨウセイ ノ ヒヘルペス セイキュウセイ ヘンエンケイ ノウエン オ トモナッタ Vogt コヤナギ ハラダビョウ ノ 1レイ

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Abstract

We report a case of Vogt-Koyanagi-Harada (VKH) disease associated with non-herpetic acute limbic encephalitis with autoantibodies against glutamate receptor ε2 in the cerebrospinal fluid. A 42-year-old woman developed a complaint of visual distortion, visual disturbance, headache and mild psychiatric symptoms, such as anxiety and depression. She was diagnosed as VKH through the fidings of fluorescein fundus angiography, which revealed patchy hypofluorescence associated with delayed choroidal filling at early fluorescein angiographic phase, and spotted choroidal hyperfluorescence and pooling of dye at late phase. Analysis of the cerebrospinal fluid (CSF) showed slight increase of leukocyte count (49/μl, mononuclear cells) and immunoglobulin (Ig) G index. An anti-GluRε2 IgM antibody was positive in CSF. Brain magnetic resonance imaging (MRI) showed a monofocal hyperintensity lesion in the left parahippocampal gyrus on T2-weighted and fluid-attenuated inversion recovery (FLAIR) images. We diagnosed her VKH disease associated with non-herpetic acute limbic encephalitis. She was treated with oral prednisone, 70mg day and her symptoms have gradually improved. To our knowledge, meningoencephalitis in VKH disease is extremely rare and the analysis of anti-GluRε2 IgM antibody in CSF has not been reported. We speculate that a certain immunologic mechanism, including the anti-GluRε2 IgM antibody, contributes to the pathogenesis of the VKH disease with non-herpetic acute limbic encephalitis.<br>

Journal

  • Rinsho Shinkeigaku

    Rinsho Shinkeigaku 49 (8), 483-487, 2009

    Societas Neurologica Japonica

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