Infliximab treatment trial in a patient with neuro-Behcet's disease unresponsive to other treatments

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  • 吃逆を主訴とし，ｉｎｆｌｉｘｉｍａｂが奏効した難治性神経ベーチェット病の１例
  • 症例報告 吃逆を主訴とし,infliximabが奏効した難治性神経ベーチェット病の1例
  • ショウレイ ホウコク キツギャク オ シュソ ト シ infliximab ガ ソウコウ シタ ナンチセイ シンケイ ベーチェットビョウ ノ 1レイ

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Abstract

A 22-year-old man with a previous uveitis episode was admitted to our hospital because of persistent hiccup. On admission, he presented right-upper quadrantanopia, mydriasis and lack of the light reflex in the left eye, left-sided hemiplegia, and bilateral pathologic hyperreflexia. The MR fluid attenuated inversion recovery images showed left side dominant, high intensity lesions on the brainstem and the diencephalon. The HLA-B51 was positive. The CSF IL-6 was extremely elevated (998pg/ml: reference value <=6.0pg/ml). Based on these, we concluded he had the neuro-Behçet's disease and treated him by high dose intravenous corticosteroids. This treatment improved his symptoms and MRI lesions, and decreased the CSF IL-6 levels initially. On 13th day after the first his discharge, however, dysarthria appeared and the CSF IL-6 levels elevated again. In addition to the high dose intravenous corticosteroids therapy for acute attack, 15mg/week of methotrexate was started to prevent the recurrence. Even with this prevention, meningitis related to neuro-Behçet's disease occurred within six weeks. We administered 5mg/kg of infliximab intravenously at 0, 2, 6, and 14 weeks. After the infliximab treatment, his symptoms improved and the IL-6 levels decreased, and no recurrence has occurred. This case supports that infliximab, anti-TNF-α agent, is a good candidate for neuro-Behçet's disease treatment when it is resistant to conventional immunosuppressive agents such as corticosteroids or methotrexate.<br>

Journal

  • Rinsho Shinkeigaku

    Rinsho Shinkeigaku 51 (4), 261-266, 2011

    Societas Neurologica Japonica

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