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首页> 外文期刊>Ophthalmic genetics >Carbonic anhydrase inhibitors in patients with X-linked retinoschisis: effects on macular morphology and function
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Carbonic anhydrase inhibitors in patients with X-linked retinoschisis: effects on macular morphology and function

机译：碳酸酐酶抑制剂患者X型视网膜肠道：对黄斑形态和功能的影响

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摘要

Background: Currently there is no medical treatment for X-linked retinoschisis (XLRS). In many retinal dystrophies, carbonic anhydrase inhibitors (CAIs) are effectively used to reduce cystoid macular edema. Prospective studies investigating the effect of CAIs in patients with XLRS are needed for the evaluation of their efficacy in this disease. The purpose of our work is to investigate the effects on macular morphology and function of oral CAIs used for the treatment of foveal lesions in patients with XLRS. Methods: Nineteen patients with a clinical diagnosis of XLRS were enrolled and prescribed oral CAIs for six months. We evaluated the therapeutic effect of CAIs with: best-corrected visual acuity (BCVA), spectral-domain optical coherence tomography, microperimetry (MP) and multifocal electroretinography (mfERG). Results: We observed a significant improvement of BCVA (p-value = 0.013), central retinal thickness (p-value = 0.004) and macular sensitivity (p-value<0.001). Moreover, in regards to mfERG responses, an increase of P1 wave amplitude was observed in three of the six rings. Conclusions: Our data supports the efficacy of oral CAIs for the treatment of macular cyst-like lesions in XLRS patients. The recovery of a normal retinal anatomy by means of oral CAIs could be useful to create the optimal circumstances for gene therapy. The increase in macular sensitivity and in P1 wave amplitude confirmed that MP and mfERG provide with an unbiased and more sensitive understanding of how macular function may respond to the use of CAIs. Therefore, we recommend the use of MP and mfERG to assess the effect of therapy in XLRS.
机译：背景：目前对X链状视网膜（XLR）没有医疗。在许多视网膜营药中，碳酸酐酶抑制剂（CAIS）有效地用于减少囊状黄斑水肿。对XLRS患者进行调查的前瞻性研究是在这种疾病中评估它们的疗效。我们的作品的目的是探讨对XLRS患者治疗心肺病变的口服CAIS的黄斑形态和功能的影响。方法：XLRS临床诊断患者六个月注册和规定口腔癌。我们评估了CAIS的治疗效果：最佳校正的视力（BCVA），光谱域光学相干性断层扫描，微观型（MP）和多焦电仪（MFRG）。结果：我们观察到BCVA的显着改善（P值= 0.013），中央视网膜厚度（P值= 0.004）和黄斑敏感性（P值<0.001）。此外，关于MFFERG响应，在六个环中的三个中观察到P1波振幅增加。结论：我们的数据支持口腔CAIS在XLRS患者中治疗黄斑囊肿状病灶的疗效。通过口服CAIS恢复正常视网膜解剖学可用于在基因治疗中创造最佳情况。黄斑灵敏度和P1波幅度的增加证实了MP和MFERG提供了对Malubul函数如何响应CAIS的响应方式的无偏见和更敏感的理解。因此，我们建议使用MP和MFERG来评估XLRS治疗的作用。

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  • 来源
    《Ophthalmic genetics》 |2019年第3期|共6页
  • 作者

    Testa Francesco; Di Iorio Valentina; Gallo Beatrice; Marchese Mario; Nesti Anna; De Rosa Giuseppe; Melillo Paolo; Simonelli Francesca;

  • 作者单位

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

    Univ Campania Luigi Vanvitelli Multidisciplinary Dept Med Surg &

    Dent Sci Eye Clin Via S Pansini;

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  • 原文格式 PDF
  • 正文语种 eng
  • 中图分类 眼科学;
  • 关键词

    Carbonic anhydrase inhibitors; multifocal electroretinogram; microperimetry; X-linked retinoschisis;

    机译：碳酸酐酶抑制剂;多灶性电气图仪;微型仪;X链状视网膜;
  • 入库时间 2022-08-20 05:21:01

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