Evidence map›Paper›PMID 39277603›Full record

ArticleScientific reports2024

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

João Pedro Marques, Nuno Ferreira, Natacha Moreno, Ana Marta, Sara Vaz-Pereira, Sérgio Estrela-Silva, José Costa, Ana Rocha Cardoso, Pedro Neves, Lilianne Duarte and 17 more

Abstract read
In one paragraph

Article in Scientific reports, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed, 1 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

7 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Guideline
  2. Review
  3. Article
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

27 authors.

João Pedro MarquesOphthalmology Department, Unidade Local de Saúde (ULS) de Coimbra, EPE, Coimbra, Portugal. marquesjoaopedro@gmail.com.
Nuno FerreiraOphthalmology Deparment, Unidade Local de Saúde (ULS) de Trás-os-Montes e Alto Douro, EPE, Vila Real, Portugal.
Natacha MorenoOphthalmology Department, Unidade Local de Saúde (ULS) de Barcelos/Esposende, EPE, Barcelos, Portugal.
Ana MartaOphthalmology Department, Unidade Local de Saúde (ULS) de Santo António, EPE, Porto, Portugal.
Sara Vaz-PereiraOphthalmology Department, Unidade Local de Saúde (ULS) de Santa Maria, EPE, Lisboa, Portugal.
Sérgio Estrela-SilvaOphthalmology Department, Unidade Local de Saúde (ULS) de São João, EPE, Porto, Portugal.
José CostaOphthalmology Department, Unidade Local de Saúde (ULS) de Braga, EPE, Braga, Portugal.
Ana Rocha CardosoOphthalmology Department, Unidade Local de Saúde (ULS) do Baixo Alentejo, EPE, Beja, Portugal.
Pedro NevesOphthalmology Department, Unidade Local de Saúde (ULS) da Arrábida, EPE, Setúbal, Portugal.
Lilianne DuarteOphthalmology Department, Unidade Local de Saúde (ULS) de Entre Douro e Vouga, EPE, Santa Maria da Feira, Portugal.
Dália MeiraOphthalmology Department, Unidade Local de Saúde (ULS) de Gaia/Espinho, EPE, Gaia, Portugal.
Joana PiresOphthalmology Department, Unidade Local de Saúde (ULS) do Alto Ave, EPE, Guimarães, Portugal.
Carlos MenezesOphthalmology Department, Unidade Local de Saúde (ULS) do Alto Minho, EPE, Viana Do Castelo, Portugal.
Filipa RodriguesOphthalmology Department, Unidade Local de Saúde (ULS) do Litoral Alentejano, EPE, Santiago Do Cacém, Portugal.
Pedro AredeOphthalmology Department, Unidade Local de Saúde (ULS) de Lisboa Ocidental, EPE, Lisboa, Portugal.
André CoutinhoOphthalmology Department, Unidade Local de Saúde (ULS) da Região de Aveiro, EPE, Aveiro, Portugal.
Diogo CabralOphthalmology Department, Unidade Local de Saúde (ULS) de Almada-Seixal, EPE, Lisboa, Portugal.
Inês CoutinhoOphthalmology Department, Unidade Local de Saúde (ULS) de Amadora/Sintra, EPE, Amadora, Portugal.
Miguel RibeiroOphthalmology Department, Unidade Local de Saúde (ULS) de Viseu Dão-Lafões, EPE, Viseu, Portugal.
Marta MacedoOphthalmology Department, Hospital Dr. Nélio Mendonça, EPE, Funchal, Portugal.
Sérgio BritoOphthalmology Department, Unidade Local de Saúde (ULS) de Castelo Branco, EPE, Castelo Branco, Portugal.
Filipe IsidroOphthalmology Department, Unidade Local de Saúde (ULS) do Algarve, EPE, Faro, Portugal.
Filipa Gomes RodriguesOphthalmology Department, Unidade Local de Saúde (ULS) do Estuário do Tejo, EPE, Vila Franca de Xira, Portugal.
João Paulo Castro SousaOphthalmology Department, Unidade Local de Saúde (ULS) da Região de Leiria, EPE, Leiria, Portugal.
Marco MarquesOphthalmology Department, Unidade Local de Saúde (ULS) do Baixo Mondego, EPE, Figueira da Foz, Portugal.
Raquel Martins, Novartis, Portugal.
Eduardo SilvaOphthalmology Department, Unidade Local de Saúde (ULS) de São José, EPE, Lisboa, Portugal.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

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.

Indexed as

Retinal DegenerationAdultFemaleGenetic TestingHealth PersonnelHumansMaleMiddle AgedPortugalPrevalenceRegistriesRetinitis PigmentosaSurveys and QuestionnairesEpidemiologyGenetic testingInherited retinal degenerationsOphthalmic geneticsRare eye diseases

Identifiers

PMID39277603
PMCPMC11401845

What OpenQuestion holds

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LicenceCC BY-NC-ND
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.