Evidence map›Paper›PMID 36980814›Full record

ArticleGenes2023

Primary Ciliary Dyskinesia in a Portuguese Bronchiectasis Outpatient Clinic.

Eduarda Milheiro Tinoco, Ana Rita Gigante, Edite Ferreira, Inês Sanches, Rute Pereira, Rosália Sá, Regina Monteiro, Mário Sousa, Ivone Pascoal

Open access · goldFull text read
In one paragraph

Article in Genes, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
1.6field-weighted citation impact, top 17% of its field
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

6 citing papers in PubMed, 6 citations in OpenAlex.

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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

9 authors at 2 institutions in 1 country.

Eduarda Milheiro TinocoDepartment of Pulmonology, Centro Hospitalar de Vila Nova de Gaia/Espinho (CHVNG/E), 4434-502 Vila Nova de Gaia, Portugal.
Ana Rita GiganteDepartment of Pulmonology, Centro Hospitalar de Vila Nova de Gaia/Espinho (CHVNG/E), 4434-502 Vila Nova de Gaia, Portugal.ORCID 0000-0002-7670-048X
Edite FerreiraUMIB-Unit for Multidisciplinary Research in Biomedicine, ITR-Laboratory for Integrative and Translational Research in Population Health, University of Porto, 4050-313 Porto, Portugal.
Inês SanchesDepartment of Pulmonology, Centro Hospitalar de Vila Nova de Gaia/Espinho (CHVNG/E), 4434-502 Vila Nova de Gaia, Portugal.ORCID 0000-0002-1664-1297
Rute PereiraUMIB-Unit for Multidisciplinary Research in Biomedicine, ITR-Laboratory for Integrative and Translational Research in Population Health, University of Porto, 4050-313 Porto, Portugal.ORCID 0000-0002-8434-5086
Rosália SáUMIB-Unit for Multidisciplinary Research in Biomedicine, ITR-Laboratory for Integrative and Translational Research in Population Health, University of Porto, 4050-313 Porto, Portugal.ORCID 0000-0002-6551-3822
Regina MonteiroDepartment of Pulmonology, Centro Hospitalar de Vila Nova de Gaia/Espinho (CHVNG/E), 4434-502 Vila Nova de Gaia, Portugal.
Mário SousaUMIB-Unit for Multidisciplinary Research in Biomedicine, ITR-Laboratory for Integrative and Translational Research in Population Health, University of Porto, 4050-313 Porto, Portugal.ORCID 0000-0002-3009-3290
Ivone PascoalDepartment of Pulmonology, Centro Hospitalar de Vila Nova de Gaia/Espinho (CHVNG/E), 4434-502 Vila Nova de Gaia, Portugal.
Universidade do Porto · PTCentro Hospitalar de Vila Nova de Gaia · PT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Primary ciliary dyskinesia (PCD) is a rare hereditary condition characterized by decreased mucociliary clearance of the airways and a compromised reproductive system, resulting in male and female infertility. Several mutations with varied clinical and pathological features have been documented, making diagnosis a challenging process. The purpose of this study is to describe the clinical and pathological features of Portuguese patients with PCD and to examine their genetic variants. A retrospective observational analysis was conducted with patients who were being monitored at a bronchiectasis outpatient clinic in 2022 and had a confirmed or high-likelihood diagnosis of PCD. In total, 17 patients were included in the study, with 12 (66.7%) having PCD confirmed and 5 (29.4%) having a high-likelihood diagnosis. Furthermore, 12 patients were subjected to transmission electron microscopy (TEM), with 7 (58.3%) exhibiting one hallmark defect. Genetic test data was obtained for all 17 patients, with 7 of them (41.2%) displaying a pathogenic/likely pathogenic mutation in homozygosity. To summarize, PCD is an uncommon but significant hereditary illness with consequences regarding morbidity and mortality. Despite the lack of a specific treatment, it is critical to confirm the diagnosis with genetic testing in order to effectively manage the disease and its accompanying disorders.

Indexed as

BronchiectasisCiliary Motility DisordersFemaleGenetic TestingHumansMalePortugalRetrospective Studiesciliary functiongeneticsnext-generation sequencingprimary ciliary dyskinesiatransmission electron microscopy

Identifiers

PMID36980814
PMCPMC10048503
OpenAlexW4321496577

What OpenQuestion holds

Textfull text, public
LicenceCC BY
measurements read27
Read underepoch 390

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.