Frederike C Oertel, Hanna G Zimmermann, Seyedamirhosein Motamedi, Charlotte Bereuter, Luca Magdalena Manthey, Fereshteh Ashtari, Rahele Kafieh, Alireza Dehghani, Mohsen Pourazizi, Lekha Pandit, Anitha D'Cunha, Orhan Aktas, Philipp Albrecht, Marius Ringelstein, Elena H Martinez-Lapiscina, Bernardo F Sanchez Dalmau, Pablo Villoslada, Nasrin Asgari, Romain Marignier, Alvaro Cobo-Calvo, Letizia Leocani, Marco Pisa, Marta Radaelli, Jacqueline Palace, Adriana Roca-Fernandez, Maria Isabel S Leite, Srilakshmi Sharma, Jerome De Seze, Thomas Senger, Michael R Yeaman, Terry J Smith, Lawrence J Cook, Alexander U Brandt, Friedemann Paul
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All groups were matched for age and sex and included from the collaborative retrospective study of retinal optical coherence tomography (OCT) in neuromyelitis optica (CROCTINO). Participants underwent OCT with central postprocessing and local neurologic examination and antibody testing. Retinal neurodegeneration was quantified as peripapillary retinal nerve fiber layer thickness (pRNFL) and combined ganglion cell and inner plexiform layer thickness (GCIPL).</p><p><strong>Results: </strong>This DN-NMOSD cohort had a history of [median (inter-quartile range)] 6 (5; 9) attacks within their 5 ± 4 years since onset. Myelitis and ON were the most common attack types. In DN-NMOSD eyes after ON, pRNFL (<i>p</i> < 0.001) and GCIPL (<i>p</i> = 0.023) were thinner compared with eyes of HCs. Even after only one ON episode, DN-NMOSD eyes already had considerable neuroaxonal loss compared with HCs. In DN-NMOSD eyes without a history of ON, pRNFL (<i>p</i> = 0.027) and GCIPL (<i>p</i> = 0.022) were also reduced compared with eyes of HCs. However, there was no difference in pRNFL and GCIPL between DN-NMOSD and AQP4-NMOSD for the whole group and for subsets with a history of ON and without a history of ON-as well as between variances of retinal layer thicknesses.</p><p><strong>Discussion: </strong>DN-NMOSD is characterized by severe retinal damage after ON and attack-independent retinal neurodegeneration. Most of the damage occurs during the first ON episode, which highlights the need for better diagnostic markers in DN-NMOSD to facilitate an earlier diagnosis as well as for effective and early treatments. 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引用次数: 0
摘要
背景和目的：系统描述双抗体血清阴性神经脊髓炎视网膜频谱疾病（DN-NMOSD）的临床表现，特别强调视网膜受累：系统描述双抗体血清阴性神经脊髓炎视网膜谱系障碍（DN-NMOSD）的临床表现，特别强调视网膜受累：本研究纳入了25名有或没有视神经炎（ON）病史的DN-NMOSD患者（48只眼）的横断面数据，以及25名水通道蛋白-4抗体血清阳性神经脊髓炎视网膜频谱障碍患者（AQP4-NMOSD，46只眼）和25名健康对照组（HCs，49只眼）的数据进行比较。所有组别在年龄和性别上都是匹配的，并纳入了神经脊髓炎视网膜光学相干断层扫描（OCT）合作回顾性研究（CROCTINO）。参试者接受了带中央后处理的光学相干断层扫描以及局部神经系统检查和抗体检测。视网膜神经变性被量化为毛周视网膜神经纤维层厚度（pRNFL）以及神经节细胞和内丛状层厚度（GCIPL）：这组 DN-NMOSD 患者在发病后的 5 ± 4 年内有 6 (5; 9) 次发作史[中位数（四分位数间距）]。脊髓炎和强直是最常见的发作类型。与普通人相比，DN-NMOSD 眼球在 ON 后的 pRNFL（p < 0.001）和 GCIPL（p = 0.023）更薄。即使只发生了一次 ON，DN-NMOSD 眼球与 HC 眼球相比已经出现了相当严重的神经轴损失。与 HCs 的眼睛相比，没有 ON 病史的 DN-NMOSD 眼睛的 pRNFL（p = 0.027）和 GCIPL（p = 0.022）也有所减少。然而，DN-NMOSD 和 AQP4-NMOSD 之间的 pRNFL 和 GCIPL 在全组、有 ON 病史和无 ON 病史的亚组以及视网膜层厚度差异之间没有差异：讨论：DN-NMOSD的特征是发生ON后视网膜的严重损伤和攻击依赖性视网膜神经变性。大部分损伤发生在第一次ON发作时，这就强调了需要更好的DN-NMOSD诊断标志物，以促进早期诊断和有效的早期治疗。在这项研究中，DN-NMOSD 患者的临床和影像学检查结果相同，这可能表明这些患者存在共同的视网膜病理。
Retinal Changes in Double-Antibody Seronegative Neuromyelitis Optica Spectrum Disorders.
Background and objectives: To systematically describe the clinical picture of double-antibody seronegative neuromyelitis optica spectrum disorders (DN-NMOSD) with specific emphasis on retinal involvement.
Methods: Cross-sectional data of 25 people with DN-NMOSD (48 eyes) with and without a history of optic neuritis (ON) were included in this study along with data from 25 people with aquaporin-4 antibody seropositive neuromyelitis optica spectrum disorder (AQP4-NMOSD, 46 eyes) and from 25 healthy controls (HCs, 49 eyes) for comparison. All groups were matched for age and sex and included from the collaborative retrospective study of retinal optical coherence tomography (OCT) in neuromyelitis optica (CROCTINO). Participants underwent OCT with central postprocessing and local neurologic examination and antibody testing. Retinal neurodegeneration was quantified as peripapillary retinal nerve fiber layer thickness (pRNFL) and combined ganglion cell and inner plexiform layer thickness (GCIPL).
Results: This DN-NMOSD cohort had a history of [median (inter-quartile range)] 6 (5; 9) attacks within their 5 ± 4 years since onset. Myelitis and ON were the most common attack types. In DN-NMOSD eyes after ON, pRNFL (p < 0.001) and GCIPL (p = 0.023) were thinner compared with eyes of HCs. Even after only one ON episode, DN-NMOSD eyes already had considerable neuroaxonal loss compared with HCs. In DN-NMOSD eyes without a history of ON, pRNFL (p = 0.027) and GCIPL (p = 0.022) were also reduced compared with eyes of HCs. However, there was no difference in pRNFL and GCIPL between DN-NMOSD and AQP4-NMOSD for the whole group and for subsets with a history of ON and without a history of ON-as well as between variances of retinal layer thicknesses.
Discussion: DN-NMOSD is characterized by severe retinal damage after ON and attack-independent retinal neurodegeneration. Most of the damage occurs during the first ON episode, which highlights the need for better diagnostic markers in DN-NMOSD to facilitate an earlier diagnosis as well as for effective and early treatments. In this study, people with DN-NMOSD presented with homogeneous clinical and imaging findings potentially suggesting a common retinal pathology in these patients.
期刊介绍：
Neurology Neuroimmunology & Neuroinflammation is an official journal of the American Academy of Neurology. Neurology: Neuroimmunology & Neuroinflammation will be the premier peer-reviewed journal in neuroimmunology and neuroinflammation. This journal publishes rigorously peer-reviewed open-access reports of original research and in-depth reviews of topics in neuroimmunology & neuroinflammation, affecting the full range of neurologic diseases including (but not limited to) Alzheimer's disease, Parkinson's disease, ALS, tauopathy, and stroke; multiple sclerosis and NMO; inflammatory peripheral nerve and muscle disease, Guillain-Barré and myasthenia gravis; nervous system infection; paraneoplastic syndromes, noninfectious encephalitides and other antibody-mediated disorders; and psychiatric and neurodevelopmental disorders. Clinical trials, instructive case reports, and small case series will also be featured.
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