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Record W4386834909 · doi:10.1186/s13073-023-01221-3

Identification of novel genetic risk factors of dilated cardiomyopathy: from canine to human

2023· article· en· W4386834909 on OpenAlexaff
Julia E. Niskanen, Åsa Ohlsson, Ingrid Ljungvall, Michaela Drögemüller, Robert F. Ernst, Dennis Dooijes, Hanneke W. M. van Deutekom, J. Peter van Tintelen, C. Snijders Blok, Marion van Vugt, Jessica van Setten, Folkert W. Asselbergs, Aleksandra Domanjko Petrič, Milla Salonen, Sruthi Hundi, Matthias Hörtenhuber, Carsten O. Daub, César L. Araujo, Ileana B. Quintero, Kaisa Kyöstilä, Maria Kaukonen, Meharji Arumilli, Riika Sarviaho, Jenni Puurunen, Sini Sulkama, Sini Karjalainen, Antti Sukura, Pernilla Syrjä, Niina Airas, Henna Pekkarinen, Ilona Kareinen, Hanna-Maaria Javela, Anna Knuuttila, Heli Nordgren, Karoliina Hagner, Tarja Pääkkönen, Antti Iivanainen, Kaarel Krjutškov, Sini Ezer, Auli Saarinen, Shintaro Katayama, Masahito Yoshihara, Abdul Kadir Mukarram, Rasha Aljelaify, Fiona Ross, Amitha Raman, Irene Stevens, Oleg Gusev, Danika L. Bannasch, Jeffrey J. Schoenebeck, Juha Kere, W. Glen Pyle, Jonas Donner, Alex V. Postma, Tosso Leeb, Göran Andersson, Marjo K. Hytönen, Jens Häggström, Maria Wiberg, Jana Friederich, Jenny Eberhard, Magdaléna Harakaľová, Frank G. van Steenbeek, Gerhard Wess, Hannes Lohi

Bibliographic record

VenueGenome Medicine · 2023
Typearticle
Languageen
FieldMedicine
TopicCardiovascular Conditions and Treatments
Canadian institutionsUniversity of GuelphSaint John Regional Hospital
FundersZonMwNational Institute for Health and Care Research
KeywordsDilated cardiomyopathyCandidate geneCardiomyopathyEtiologyTranscriptomeMedicineHeart failureHuman geneticsGenome-wide association studyHeart diseasePhenotypeDiseaseBioinformaticsCardiologyInternal medicineGeneBiologyGeneticsGene expressionSingle-nucleotide polymorphism

Abstract

fetched live from OpenAlex

BACKGROUND: Dilated cardiomyopathy (DCM) is a life-threatening heart disease and a common cause of heart failure due to systolic dysfunction and subsequent left or biventricular dilatation. A significant number of cases have a genetic etiology; however, as a complex disease, the exact genetic risk factors are largely unknown, and many patients remain without a molecular diagnosis. METHODS: We performed GWAS followed by whole-genome, transcriptome, and immunohistochemical analyses in a spontaneously occurring canine model of DCM. Canine gene discovery was followed up in three human DCM cohorts. RESULTS: Our results revealed two independent additive loci associated with the typical DCM phenotype comprising left ventricular systolic dysfunction and dilatation. We highlight two novel candidate genes, RNF207 and PRKAA2, known for their involvement in cardiac action potentials, energy homeostasis, and morphology. We further illustrate the distinct genetic etiologies underlying the typical DCM phenotype and ventricular premature contractions. Finally, we followed up on the canine discoveries in human DCM patients and discovered candidate variants in our two novel genes. CONCLUSIONS: Collectively, our study yields insight into the molecular pathophysiology of DCM and provides a large animal model for preclinical studies.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.001
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0020.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.026
GPT teacher head0.280
Teacher spread0.254 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations14
Published2023
Admission routes1
Has abstractyes

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