A case of paraneoplastic optic neuropathy and outer retinitis positive for autoantibodies against collapsin response mediator protein-5, recoverin, and α-enolase.
A case of paraneoplastic optic neuropathy and outer retinitis positive for autoantibodies against collapsin response mediator protein-5, recoverin, and α-enolase.
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DOI：
10.1186/1471-2415-14-5
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发表时间：
2014-01-16
影响因子：
2
通讯作者：
Ishida S
中科院分区：
文献类型：
--
作者：
Saito M;Saito W;Kanda A;Ohguro H;Ishida S
已知针对恢复蛋白或塌陷蛋白反应介导蛋白 (CRMP)-5 的特异性交叉反应自身免疫会分别导致癌症相关的视网膜病变或副肿瘤性视神经病变。我们报告了一个罕见的小细胞肺癌病例，这些病例出现双侧神经视网膜炎和单侧局灶性外视网膜炎，这些抗体呈阳性。一名 67 岁男性出现双侧神经视网膜炎，右眼黄斑中心凹渗出。光学相干断层扫描显示右眼中央凹处有一个从内核层延伸到感光层的圆顶状高反射病变。单次闪光视网膜电图显示双眼 a 波正常，左眼 b 波略有减少。感染血清学筛查测试结果在正常范围内。口服泼尼松龙后，患者视盘肿胀和黄斑渗出迅速改善。系统筛查发现肺小细胞癌并开始全身化疗。使用患者血清进行的免疫印迹分析检测到针对恢复素、CRMP-5 和 α-烯醇化酶的自身抗体，但未检测到针对碳酸酐酶 II 的自身抗体。神经视网膜炎曾在影像学上的癌症几乎缓解后消退，但在癌症复发后又复发。该具有抗视网膜和抗视神经抗体的癌症患者的神经视网膜炎的发展很大程度上取决于癌症的活动性，这表明可能涉及副肿瘤机制。患有副肿瘤性视神经病变和视网膜病变的患者可能会对多种抗原产生自身免疫反应，从而导致各种眼科受累。
Specific cross-reacting autoimmunity against recoverin or collapsin response mediator protein (CRMP)-5 is known to cause cancer-associated retinopathy or paraneoplastic optic neuropathy, respectively. We report a rare case with small cell lung carcinoma developing bilateral neuroretinitis and unilateral focal outer retinitis positive for these antibodies. A 67-year-old man developed bilateral neuroretinitis and foveal exudation in the right eye. Optical coherence tomography showed a dome-shaped hyperreflective lesion extending from inner nuclear layer to the photoreceptor layer at the fovea in the right eye. Single-flash electroretinography showed normal a-waves in both eyes and slightly reduced b-wave in the left eye. Results of serological screening tests for infection were within normal limits. The patient’s optic disc swelling and macular exudation rapidly improved after oral administration of prednisolone. Systemic screening detected lung small cell carcinoma and systemic chemotherapy was initiated. Immunoblot analyses using the patient’s serum detected autoantibodies against recoverin, CRMP-5, and α-enolase, but not carbonic anhydrase II. Neuroretinitis once resolved after almost remission of carcinoma on imaging but it recurred following the recurrence of carcinoma. The development of neuroretinitis in this cancer patient with anti-retinal and anti-optic nerve antibodies depended largely on the cancer activity, suggesting the possible involvement of paraneoplastic mechanisms. Patients with paraneoplastic optic neuropathy and retinopathy are likely to develop autoimmune responses against several antigens, thus leading to various ophthalmic involvements.
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