Evidence map›Paper›PMID 36004757›Full record

ArticleAnimal genetics2022

KRT5 missense variant in a Cardigan Welsh Corgi with epidermolysis bullosa simplex.

Sarah Kiener, Elizabeth A Mauldin, Vidhya Jagannathan, Margret L Casal, Tosso Leeb

Open access · hybridAbstract read
In one paragraph

Article in Animal genetics, 2022. 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
0.6field-weighted citation impact, top 39% 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, 5 citations in OpenAlex.

  1. IndependentGenes · 2023
    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

5 authors at 2 institutions in 2 countries.

Sarah KienerVetsuisse Faculty, Institute of Genetics, University of Bern, Bern, Switzerland.ORCID https://orcid.org/0000-0002-2714-2370
Elizabeth A MauldinSchool of Veterinary Medicine, University of Pennsylvania, Philadelphia, Pennsylvania, USA.ORCID https://orcid.org/0000-0002-4718-6071
Vidhya JagannathanVetsuisse Faculty, Institute of Genetics, University of Bern, Bern, Switzerland.ORCID https://orcid.org/0000-0002-8155-0041
Margret L CasalSchool of Veterinary Medicine, University of Pennsylvania, Philadelphia, Pennsylvania, USA.ORCID https://orcid.org/0000-0002-1224-6708
Tosso LeebVetsuisse Faculty, Institute of Genetics, University of Bern, Bern, Switzerland.ORCID https://orcid.org/0000-0003-0553-4880
University of Bern · CHUniversity of Pennsylvania · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Epidermolysis bullosa (EB) is a group of blistering disorders that includes several subtypes, classified according to their level of cleavage. Typical clinical signs are blisters and erosions resulting from minimal trauma. The disease has been described in many mammalian species and pathogenic variants in at least 18 different genes have been identified. In the present study, we investigated a Cardigan Welsh Corgi with congenital clinical signs consistent with epidermolysis bullosa. The puppy had blisters and erosions on the paw pads, and the oral mucosa. Histologic examination demonstrated the typical clefting between the dermis and epidermis and confirmed the clinical suspicion. We obtained whole genome sequencing data from the affected puppy and searched for variants in candidate genes known to cause EB. This revealed a heterozygous missense variant, KRT5:p.(E476K), affecting the highly conserved KLLEGE motif of keratin 5. The mutant allele in the affected puppy arose owing to a de novo mutation event as it was absent from both unaffected parents. Knowledge of the functional impact of KRT5 variants in other species together with the demonstration of the de novo mutation event establishes KRT5:p.(E476K) as causative variant for the observed EBS.

Indexed as

Dog DiseasesEpidermolysis Bullosa SimplexAnimalsBlisterDogsKeratin-14Keratin-5MammalsMutation, MissenseKeratin-14Keratin-5animal modelCanis lupus familiarisdermatologydoggenodermatosisprecision medicineskinveterinary medicine

Identifiers

PMID36004757
PMCPMC9804678
OpenAlexW4293090152

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.