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葡萄牙遗传性视网膜变性的现行管理(IRD-PT 调查)。

Current management of inherited retinal degenerations in Portugal (IRD-PT survey).

机构信息

Ophthalmology Department, Unidade Local de Saúde (ULS) de Coimbra, EPE, Coimbra, Portugal.

Ophthalmology Department, Unidade Local de Saúde (ULS) de Coimbra, EPE, Praceta Prof. Mota Pinto, 3000-075, Coimbra, Portugal.

出版信息

Sci Rep. 2024 Sep 14;14(1):21473. doi: 10.1038/s41598-024-72589-4.

DOI:10.1038/s41598-024-72589-4
PMID:39277603
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11401845/
Abstract

Inherited retinal dystrophies/degenerations (IRDs) are the leading cause of visual impairment and incurable familial blindness in the Western world. Given the clinical and genetic heterogeneity, establishing a molecular diagnosis is especially relevant. The aim of this study was to perform the first nationwide survey to understand the prevalence and current management of IRDs in Portugal. A response was obtained from 26 healthcare providers (HCP) (76.5% response rate). Only 4 respondents reported not managing IRD patients. Most HCPs (68.1%) reported managing up to 100 patients, while three currently manage between 501 and 1000 patients. Based on the Portuguese population, an estimated IRD prevalence of 0.031%, i.e., about 1 in 3000 individuals, was calculated. In most HCPs (86.3%), most patients are adults, and non-syndromic retinitis pigmentosa is the most frequent diagnosis. Only 4 HCPs currently use the national, web-based IRD registry (IRD-PT). However, all but one respondent expressed interest in participating in such a registry. Genetic testing is available in 54.5%, with 58.3% HCPs reporting solved rates between 61-80%, but 4 to 9 months to get a genetic test result in 83.4% of cases. Based on this survey, the prevalence of biallelic RPE65-associated disease in Portugal is 0.00031%, i.e., approximately 1:300,000 individuals. Data from this study provide vital background information on national differences in the diagnosis and management of IRD patients. Nationwide implementation of the IRD-PT registry should be encouraged and supported to provide population-based reference data and to identify patients eligible for current and future therapies.

摘要

遗传性视网膜病变/变性(IRDs)是西方世界导致视力损害和不可治愈的家族性失明的主要原因。鉴于临床和遗传的异质性,建立分子诊断尤为重要。本研究的目的是进行首次全国性调查,以了解葡萄牙 IRD 的流行情况和当前管理情况。共有 26 名医疗保健提供者(HCP)(76.5%的回复率)做出了回应。只有 4 名受访者报告未管理 IRD 患者。大多数 HCP(68.1%)报告管理多达 100 名患者,而 3 名目前管理 501 至 1000 名患者。根据葡萄牙人口,估计 IRD 的患病率为 0.031%,即每 3000 人中有 1 人。在大多数 HCP(86.3%)中,大多数患者是成年人,非综合征性色素性视网膜炎是最常见的诊断。目前只有 4 名 HCP 使用国家网络 IRD 登记处(IRD-PT)。然而,除了一名受访者外,所有受访者都表示有兴趣参与这样的登记处。54.5%的人可进行基因检测,58.3%的 HCP 报告解决率在 61-80%之间,但在 83.4%的情况下,基因检测结果需要 4 至 9 个月才能得出。根据这项调查,葡萄牙双等位基因 RPE65 相关疾病的患病率为 0.00031%,即大约每 30 万人中有 1 人。本研究的数据提供了关于 IRD 患者诊断和管理的国家差异的重要背景信息。应鼓励并支持在全国范围内实施 IRD-PT 登记处,以提供基于人群的参考数据,并确定有资格接受当前和未来治疗的患者。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/ae5f33c64c1f/41598_2024_72589_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/b894144c9827/41598_2024_72589_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/7d3a7c0b8833/41598_2024_72589_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/66e1993dd799/41598_2024_72589_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/babef752eeb5/41598_2024_72589_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/ae5f33c64c1f/41598_2024_72589_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/b894144c9827/41598_2024_72589_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/7d3a7c0b8833/41598_2024_72589_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/66e1993dd799/41598_2024_72589_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/babef752eeb5/41598_2024_72589_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bd34/11401845/ae5f33c64c1f/41598_2024_72589_Fig5_HTML.jpg

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