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Longitudinal optic neuritis-unrelated visual evoked potential changes in NMO spectrum disorders.

NMO谱系障碍中的纵向视神经炎-无关视觉诱发电位变化。

  • 影响因子:3.85
  • DOI:10.1212/WNL.0000000000008684
  • 作者列表:"Ringelstein M","Harmel J","Zimmermann H","Brandt AU","Paul F","Haarmann A","Buttmann M","Hümmert MW","Trebst C","Schroeder C","Ayzenberg I","Kleiter I","Hellwig K","Havla J","Kümpfel T","Jarius S","Wildemann B","Rommer P","Weber MS","Pellkofer H","Röpke L","Geis C","Retzlaff N","Zettl U","Deppe M","Klotz L","Young K","Stellmann JP","Kaste M","Kermer P","Marouf W","Lauda F","Tumani H","Graf J","Klistorner A","Hartung HP","Aktas O","Albrecht P","Neuromyelitis Optica Study Group (NEMOS).
  • 发表时间:2020-01-28
Abstract

OBJECTIVE:To investigate if patients with neuromyelitis optica spectrum disorder (NMOSD) develop subclinical visual pathway impairment independent of acute attacks. METHODS:A total of 548 longitudinally assessed full-field visual evoked potentials (VEP) of 167 patients with NMOSD from 16 centers were retrospectively evaluated for changes of P100 latencies and P100-N140 amplitudes. Rates of change in latencies (RCL) and amplitudes (RCA) over time were analyzed for each individual eye using linear regression and compared using generalized estimating equation models. RESULTS:The rates of change in the absence of optic neuritis (ON) for minimal VEP intervals of ≥3 months between baseline and last follow-up were +1.951 ms/y (n = 101 eyes; SD = 6.274; p = 0.012) for the P100 latencies and -2.149 µV/y (n = 64 eyes; SD = 5.013; p = 0.005) for the P100-N140 amplitudes. For minimal VEP intervals of ≥12 months, the RCL was +1.768 ms/y (n = 59 eyes; SD = 4.558; p = 0.024) and the RCA was -0.527 µV/y (n = 44 eyes; SD = 2.123; p = 0.111). The history of a previous ON >6 months before baseline VEP had no influence on RCL and RCA. ONs during the observational period led to mean RCL and RCA of +11.689 ms/y (n = 16 eyes; SD = 17.593; p = 0.003) and -1.238 µV/y (n = 11 eyes; SD = 3.708; p = 0.308), respectively. CONCLUSION:This first longitudinal VEP study of patients with NMOSD provides evidence of progressive VEP latency delay occurring independently of acute ON. Prospective longitudinal studies are needed to corroborate these findings and help to interpret the clinical relevance.

摘要

目的: 探讨视神经脊髓炎谱系障碍 (NMOSD) 患者是否发生独立于急性发作的亚临床视觉通路损害。 方法: 对来自 16 个中心的 548 例NMOSD患者的 167 个纵向评估的全场视觉诱发电位 (VEP) 进行回顾性评估,以了解P100 潜伏期和P100-N140 波幅的变化。使用线性回归分析每只眼睛的潜伏期 (RCL) 和振幅 (RCA) 随时间的变化率,并使用广义估计方程模型进行比较。 结果: 无视神经炎的变化率 (ON) 在基线和最后一次随访之间 ≥ 3 个月的最小VEP间隔为 + 1.951 ms/y (n = 101 只眼; SD = 6.274; p = 0.012) 对于P100 潜伏期和-2.149 µ v/y (n = 64 只眼; SD = 5.013;P = 0.005) 的P100-N140 振幅。对于 ≥ 12 个月的最小VEP间隔,RCL为 + 1.768 ms/y (n = 59 眼; SD = 4.558; p = 0.024) RCA为-0.527 µ v/y (n = 44 只眼; SD = 2.123; p = 0.111)。基线VEP前> 6 个月的病史对RCL和RCA无影响。观察期间的ONs导致平均RCL和RCA为 + 11.689 ms/y (n = 16 眼; SD = 17.593; p = 0.003) 和-1.238 µ v/y (n = 11 只眼; SD = 3.708; p = 0.308)。 结论: 这项首次对NMOSD患者进行的纵向VEP研究提供了独立于急性ON发生的进行性VEP潜伏期延迟的证据。需要前瞻性纵向研究来证实这些发现,并帮助解释临床相关性。

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影响因子:1.52
发表时间:2020-04-02
DOI:10.1080/09273948.2019.1597896
作者列表:["Apivatthakakul A","Kunavisarut P","Rothova A","Pathanapitoon K"]

METHODS::Purpose: To report on ocular Vogt-Koyanagi-Harada (VKH)-like syndrome under vemurafenib treatment for metastatic melanoma.Design: A case report.Method: Description of clinical and imaging manifestations including fundus photography, fluorescein, and indocyanine green angiography.Results: A 46-year-old Thai female was diagnosed with metastatic melanoma of the skin and had been treated with multiple surgical excisions, radiotherapy, and vemurafenib (initial dose 480 mg orally twice daily, subsequently increased to maximum dose of 960 mg twice daily). After 6 months of vemurafenib use, she complained of bilateral redness and photophobia and was diagnosed with bilateral anterior uveitis, which was topically treated. Two weeks later, her visual acuity (VA) sharply deteriorated to 20/80 and counting fingers. Ocular examination at that stage stronly resembled acute VKH disease. She exhibited intraocular inflammation, and her fundus examination revealed bilateral optic disc swelling and serous retinal detachment. Fluorescein angiogram showed disc leakage and multiple pinpoint hyperfluorescence leakage spots and indocyanine green demonstrated multiple hypofluorescent spots. Oral prednisolone 30 mg/day was commenced while vemurafenib medication was ceased. Three weeks later, her vision improved, and serous retinal detachment subsided. However, her cutaneous melanoma recurred.Conclusions: Vemurafenib, a potential adjunct treatment for metastatic melanoma, was complicated by the development of panuveitis, papillitis, and multiple serous detachments. These ocular symptoms were similar to the presentation of acute VKH syndrome.

翻译标题与摘要 下载文献
影响因子:2.19
发表时间:2020-01-01
DOI:10.1111/dmcn.14268
作者列表:["Crow YJ","Shetty J","Livingston JH"]

METHODS::Comprehensive reviews of the clinical characteristics and pathogenesis of Aicardi-Goutières syndrome (AGS), particularly its contextualization within a putative type I interferonopathy framework, already exist. However, recent reports of attempts at treatment suggest that an assessment of the field from a therapeutic perspective is warranted at this time. Here, we briefly summarize the neurological phenotypes associated with mutations in the seven genes so far associated with AGS, rehearse current knowledge of the pathology as it relates to possible treatment approaches, critically appraise the potential utility of therapies, and discuss the challenges in assessing clinical efficacy. WHAT THIS PAPER ADDS: Progress in understanding AGS disease pathogenesis has led to the first attempts at targeted treatment. Further rational therapies are expected to become available in the short- to medium-term.

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翻译标题与摘要 下载文献
影响因子:1.52
发表时间:2020-04-02
DOI:10.1080/09273948.2019.1603312
作者列表:["Takayama K","Obata H","Takeuchi M"]

METHODS::Purpose: To report the efficacy of adalimumab in a case of chronic Vogt-Koyanagi-Harada (VKH) disease refractory to conventional corticosteroids and immunosuppressive therapy and complicated by central serous chorioretinopathy (CSC).Case report: A 66-year-old woman diagnosed with VKH was treated with intravenous corticosteroids followed by oral corticosteroids and cyclosporine. However, systemic corticosteroids could not be tapered because of recurrent ocular inflammation and systemic complications (diabetes mellitus, moon face, bone weakness), while CSC appeared in both eyes. A diagnosis of chronic VKH resistant to medications complicated by corticosteroid-induced CSC was made. Systemic corticosteroids and cyclosporine were tapered and adalimumab initiated. Bilateral ocular inflammation and CSC were gradually reduced and visual acuity improved without any adverse effect. Twelve months after starting adalimumab monotherapy, no signs of active VKH and CSC were present.Conclusions: Adalimumab is one of the effective therapeutic options for refractory VKH disease complicated with corticosteroid-induced adverse effects.

神经系统自身免疫性疾病方向

神经系统自身免疫性疾病是以自身免疫细胞、免疫分子等攻击神经系统为主要致病机制的自身免疫性疾病。在免疫反应中,作用于神经系统自身抗原的致病抗体统称为神经系统自身抗体。

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