Evidence map›Paper›PMID 22446493›Full record

ArticleMolecular and cellular probes2012

Missense mutation in PFKM associated with muscle-type phosphofructokinase deficiency in the Wachtelhund dog.

G Inal Gultekin, K Raj, S Lehman, A Hillström, U Giger

Abstract read
In one paragraph

Article in Molecular and cellular probes, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed
0.4field-weighted citation impact, top 31% 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

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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

11 citing papers in PubMed, 15 citations in OpenAlex.

  1. Article
  2. Article
  3. Gene therapy for glycogen storage diseases.Journal of inherited metabolic disease · 2024
    Review
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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

5 authors at 2 institutions in 2 countries.

G Inal GultekinSection of Medical Genetics, University of Pennsylvania, Philadelphia, PA, USA.
K Raj
S Lehman
A Hillström
U Giger
University of Pennsylvania · USSwedish University of Agricultural Sciences · SE

Funding

X-LINKED SEVERE COMBINED IMMUNODEFICIENCY IN THE DOGP40RR002512 · NCRR · UNIVERSITY OF PENNSYLVANIA · PI HASKINS, MARK E · 1985 to 2011
$7.6M
Short-term Training: students in health professional schoolsT35OD010919 · OD · UNIVERSITY OF PENNSYLVANIA · PI Michael Lee Atchison · 2012 to 2026
$1.8M
NCRR NIH HHS P40 RR002512NCRR NIH HHS RR002512NIH HHS T35 OD010919
6 · The paper itself

Abstract

Hereditary muscle-type phosphofructokinase (PFK) deficiency causing intermittent hemolytic anemia and exertional myopathy due to a single nonsense mutation in PFKM has been previously described in English Springer and American Cocker Spaniels, Whippets, and mixed breed dogs. We report here on a new missense mutation associated with PFK deficiency in Wachtelhunds. Coding regions of the PFKM gene were amplified from genomic DNA and/or cDNA reverse-transcribed from RNA of EDTA blood of PFK-deficient and clinically healthy Wachtelhunds and control dogs. The amplicons were sequenced and compared to the published canine PFKM sequence. A point mutation (c.550C>T, in the coding sequence of PFKM expressed in blood) was found in all 4 affected Wachtelhunds. This missense mutation results in an amino acid substitution of arginine (Arg) to tryptophan (Trp) at position 184 of the protein expressed in blood (p.Arg184Trp). The mutation is located within an alpha-helix, and based on the SIFT analysis, this amino acid substitution is not tolerated. Amplifying the region around this mutation and digesting the PCR fragment with the restriction enzyme MspI, produces fragments that readily differentiate between PFK-deficient, carrier, and normal animals. Furthermore, we document 2 additional upstream PFKM exons expressed in canine testis but not in blood. Despite their similar phenotypic appearance and use for hunting, Wachtelhunds and English Springer Spaniels are not thought to have common ancestors. Thus, it is not surprising that different mutations are responsible for PFK deficiency in these breeds. Knowledge of the molecular basis of PFK deficiency in Wachtelhunds provides an opportunity to screen and control the spread of this deleterious trait.

Indexed as

Mutation, MissenseAmino Acid SequenceAnimalsBase SequenceConserved SequenceDNA Mutational AnalysisDog DiseasesDogsFemaleGenetic Association StudiesGlycogen Storage Disease Type VIIMaleMolecular Sequence DataPedigreePhosphofructokinasesPhosphofructokinases

Identifiers

PMID22446493
PMCPMC3485442
OpenAlexW2063101732

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Registered trials

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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.