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Genetic Mutations in Familial Pulmonary Fibrosis: A Next Generation Sequencing (NGS) Study.

2025· article· W4416638986 on OpenAlexaff
Simone Montini, Vincenzo Alfredo Marando, Sara Lettieri, Mariachiara Crescenzi, A. Ripamonti, Lucrezia Pisanu, Francesco Rocco Bertuccio, Simone Cordoni, Davide Piloni, Francesca Mariani, Angelo Guido Corsico, Ilaria Campo

Bibliographic record

Venuenot available
Typearticle
Language
FieldMedicine
TopicInterstitial Lung Diseases and Idiopathic Pulmonary Fibrosis
Canadian institutionsUniversity Hospital Foundation
Fundersnot available
KeywordsProbandMutationPulmonary fibrosisIdiopathic pulmonary fibrosisGenetic testingGeneFamily historyHaplotype

Abstract

fetched live from OpenAlex

Introduction. Familial Pulmonary Fibrosis (FPF) occurs when two or more family members are affected by Idiopathic Pulmonary Fibrosis (IPF) or any form of Idiopathic Interstitial Pneumonia (IIP). FPF is often underdiagnosed, particularly in patients without a clear family history or with nonspecific early-stage symptoms. Early identification of genetic mutations linked to pulmonary fibrosis is crucial but raises ethical concerns, especially given the lack of curative treatment. Aim. To identify genetic mutations associated with FPF in a family with multiple affected members. Methods. A NGS gene panel targeting genes involved in pulmonary fibrosis was designed and tested at the Pneumology Unit of IRCCS Policlinico San Matteo Hospital on a family with three first-degree relatives affected by pulmonary fibrosis. Results. Identified mutations included heterozygous: SLC7A7 c.1417C>T, TGFBR2 c.458del, TERT c.2608T>G. The TERT mutation was present in both the proband (a female with IPF features) and her brother (who showed IPF features). Another brother died from unspecified pulmonary fibrosis. The SLC7A7, TGFBR2 and TERT mutations were found in both the proband and her niece, who was healthy. Comprehensive radiological studies were performed on all affected family members, and patients with IPF features were treated with antifibrotic agents. Discussion. The SLC7A7 mutation has been associated with lysinuric protein intolerance, TGFBR2 with cardiovascular and neurocognitive syndromes, and the TERT mutation (c.2608T>G) is novel. While these mutations have not been previously linked to FPF, their combined presence in one individual may suggest a potential pathogenic relationship.

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)0.000
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: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.007

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0010.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.000
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 head0.037
GPT teacher head0.287
Teacher spread0.250 · 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 designObservational
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 routes1
Has abstractyes

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