Evidence map›Paper›PMID 31593572›Full record

ArticlePloS one2019

Two novel and correlated CF-causing insertions in the (TG)mTn tract of the CFTR gene.

Silvia Pierandrei, Giovanna Blaconà, Benedetta Fabrizzi, Giuseppe Cimino, Natalia Cirilli, Nicole Caporelli, Antonio Angeloni, Marco Cipolli, Marco Lucarelli

Open access · goldAbstract read
In one paragraph

Article in PloS one, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.3field-weighted citation impact, top 33% 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

2 citing papers in PubMed, 3 citations in OpenAlex.

  1. Theratyping cystic fibrosisThe European respiratory journal · 2021
    Article
  2. Article
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 3 institutions in 1 country.

Silvia PierandreiDept. of Mother-Child and Urologic Sciences, Sapienza University of Rome, Rome, Italy.
Giovanna BlaconàDept. of Experimental Medicine, Sapienza University of Rome, Rome, Italy.
Benedetta FabrizziCystic Fibrosis Care Center, Mother - Child Department, United Hospitals, Ancona, Italy.
Giuseppe CiminoCystic Fibrosis Care Center, Umberto I Hospital, Rome, Italy.
Natalia CirilliCystic Fibrosis Care Center, Mother - Child Department, United Hospitals, Ancona, Italy.
Nicole CaporelliCystic Fibrosis Care Center, Mother - Child Department, United Hospitals, Ancona, Italy.
Antonio AngeloniDept. of Experimental Medicine, Sapienza University of Rome, Rome, Italy.
Marco CipolliCystic Fibrosis Care Center, Mother - Child Department, United Hospitals, Ancona, Italy.
Marco LucarelliDept. of Experimental Medicine, Sapienza University of Rome, Rome, Italy.ORCID 0000-0002-5538-2494
Sapienza University of Rome · ITIstituto Pasteur · ITPoliclinico Umberto I · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Two novel and related pathogenic variants of the Cystic Fibrosis Transmembrane conductance Regulator (CFTR) gene were structurally and functionally characterized. These alterations have not been previously described in literature. Two patients with diagnosis of Cystic Fibrosis (CF) based on the presence of one mutated allele, p.Phe508del, pathological sweat test and clinical symptoms were studied. To complete the genotypes of both patients, an extensive genetic and functional analysis of the CFTR gene was performed. Extensive genetic characterization confirmed the presence of p.Phe508del pathogenic variant and revealed, in both patients, the presence of an insertion of part of intron 10 in intron 9 of the CFTR gene, within the (TG)m repeat, with a variable poly-T stretch. The molecular lesions resulted to be very similar in both patients, with only a difference in the number of T in the poly-T stretch. The functional characterization at RNA level revealed a complete anomalous splicing, without exon 10, from the allele with the insertion of both patients. Consequently, the alleles with the insertions are expected not to contribute to the formation of a functional CFTR protein. Molecular and functional features of these alterations are compatible with the definition of novel CF-causing variants of the CFTR gene. This also allowed the completion of the genetic characterization of both patients.

Indexed as

AdolescentAllelesBase SequenceChildCystic FibrosisCystic Fibrosis Transmembrane Conductance RegulatorExonsFemaleGenotypeHumansIntronsMaleMutagenesis, InsertionalMutationRNA SplicingCFTR protein, humanCystic Fibrosis Transmembrane Conductance Regulator

Identifiers

PMID31593572
PMCPMC6782095
OpenAlexW2979403232

What OpenQuestion holds

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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.