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Record W4417223547 · doi:10.1016/j.cjcpc.2025.11.003

Functional Pulmonary Atresia With Overlapping Dilated, Hypertrophic, and Excessive Trabeculation Cardiomyopathy in a Neonate With a TPM1 Mutation

2025· article· en· W4417223547 on OpenAlexafffund
Erika Correia, Anaïs Medouni, Robert S. Tuta, Chloé Wanert, Marie‐Josée Raboisson, Grégor Andelfinger

Bibliographic record

VenueCJC Pediatric and Congenital Heart Disease · 2025
Typearticle
Languageen
FieldMedicine
TopicCardiomyopathy and Myosin Studies
Canadian institutionsCentre Hospitalier Universitaire Sainte-Justine
FundersCanadian Institutes of Health ResearchCHU Sainte-Justine Foundation
KeywordsCardiomyopathyMutationHeart diseasePulmonary atresiaHeart failure

Abstract

fetched live from OpenAlex

Cardiomyopathies in neonates represent a heterogeneous group of disorders with diverse genetic underpinnings and phenotypic manifestations.Among sarcomeric protein mutations, TPM1, encoding a-tropomyosin, is well recognized as a causal gene for hypertrophic cardiomyopathy, dilated cardiomyopathy, excessive trabeculation cardiomyopathy (LVET), and, less frequently, restrictive cardiomyopathy.Here, we report a neonate with a TPM1 missense mutation who presented prenatally with functional pulmonary atresia and a unique combination of cardiomyopathy phenotypes.Despite the concerning presentation, the infant evolved favorably with supportive management, highlighting the variability of clinical trajectories in this condition. CaseA 26-year-old gravida 3, para 0 woman was referred at 24 + 6 weeks of gestation for suspected congenital heart disease on routine ultrasound.Fetal echocardiography was remarkable for a morphologically normal tricuspid and mildly thickened pulmonary valve.Antegrade pulmonary flow was absent, and the right ventricle was dilated and hypertrophied with decreased systolic function.A diagnosis of pulmonary valve atresia with intact ventricular septum was made initially.No extracardiac anomalies were identified.Amniocentesis for genetic investigations was offered but declined.The family history was noncontributory.The fetal echocardiographic

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)0.006
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.006
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0020.001
Science and technology studies0.0020.002
Scholarly communication0.0010.001
Open science0.0010.002
Research integrity0.0030.002
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 head0.006
GPT teacher head0.215
Teacher spread0.209 · 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 designCase report
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

Citations0
Published2025
Admission routes2
Has abstractno

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