Evidence map›Paper›PMID 26330336›Full record

ArticleJournal of the American Heart Association2015

Toward Personalized Medicine: Using Cardiomyocytes Differentiated From Urine-Derived Pluripotent Stem Cells to Recapitulate Electrophysiological Characteristics of Type 2 Long QT Syndrome.

Mariam Jouni, Karim Si-Tayeb, Zeineb Es-Salah-Lamoureux, Xenia Latypova, Benoite Champon, Amandine Caillaud, Anais Rungoat, Flavien Charpentier, Gildas Loussouarn, Isabelle Baró and 3 more

Open access · goldAbstract readValidation Study
PubMed Full text at PMC Publisher
In one paragraph

Article in Journal of the American Heart Association, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers.

0numbers the graph read from it
0cells of the map it votes in
43citing papers in PubMed
2.6field-weighted citation impact, top 10% 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

43 citing papers in PubMed, 71 citations in OpenAlex.

  1. Review
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  6. Synthetic embryology of the human heart.Frontiers in cell and developmental biology · 2024
    Review
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  18. Long QT SyndromeCirculation. Arrhythmia and electrophysiology · 2021
    Article
  19. 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

13 authors at 5 institutions in 2 countries.

Mariam JouniInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) ER045, PRASE, Laboratory of Stem Cells, Lebanese University, Beirut, Lebanon (M.J., K.Z.).
Karim Si-TayebInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Zeineb Es-Salah-LamoureuxInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Xenia LatypovaInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Benoite ChamponInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Amandine CaillaudInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Anais RungoatInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Flavien CharpentierInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Gildas LoussouarnInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Isabelle BaróInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Kazem ZibaraER045, PRASE, Laboratory of Stem Cells, Lebanese University, Beirut, Lebanon (M.J., K.Z.).
Patricia LemarchandInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Nathalie GaboritInserm, UMR 1087, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CNRS, UMR 6291, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) Université de Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.) CHU Nantes, l'institut du thorax, Nantes, France (M.J., K.S.T., Z.E.S.L., X.L., B.C., A.C., A.R., F.C., G.L., I.B., P.L., N.G.).
Centre National de la Recherche Scientifique · FRInstitut du Thorax · FRNantes Université · FRInserm · FRLebanese University · LB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundHuman genetically inherited cardiac diseases have been studied mainly in heterologous systems or animal models, independent of patients' genetic backgrounds. Because sources of human cardiomyocytes (CMs) are extremely limited, the use of urine samples to generate induced pluripotent stem cell-derived CMs would be a noninvasive method to identify cardiac dysfunctions that lead to pathologies within patients' specific genetic backgrounds. The objective was to validate the use of CMs differentiated from urine-derived human induced pluripotent stem (UhiPS) cells as a new cellular model for studying patients' specific arrhythmia mechanisms. METHODS AND

resultsCells obtained from urine samples of a patient with long QT syndrome who harbored the HERG A561P gene mutation and his asymptomatic noncarrier mother were reprogrammed using the episomal-based method. UhiPS cells were then differentiated into CMs using the matrix sandwich method.UhiPS-CMs showed proper expression of atrial and ventricular myofilament proteins and ion channels. They were electrically functional, with nodal-, atrial- and ventricular-like action potentials recorded using high-throughput optical and patch-clamp techniques. Comparison of HERG expression from the patient's UhiPS-CMs to the mother's UhiPS-CMs showed that the mutation led to a trafficking defect that resulted in reduced delayed rectifier K(+) current (IKr). This phenotype gave rise to action potential prolongation and arrhythmias.

conclusionsUhiPS cells from patients carrying ion channel mutations can be used as novel tools to differentiate functional CMs that recapitulate cardiac arrhythmia phenotypes.

Indexed as

Cell DifferentiationAction PotentialsCell Culture TechniquesCells, CulturedCellular Reprogramming TechniquesElectrocardiographyERG1 Potassium ChannelEther-A-Go-Go Potassium ChannelsFemaleGenetic Predisposition to DiseaseHigh-Throughput Screening AssaysHumansLong QT SyndromeMaleMiddle AgedMutation, MissenseERG1 Potassium ChannelEther-A-Go-Go Potassium ChannelsKCNH2 protein, humanarrhythmiacardiomyocytesHERG genelong QT syndromeurine‐derived induced pluripotent stem cells

Identifiers

PMID26330336
PMCPMC4599503
OpenAlexW2152299286

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