MétaCan
Menu
Back to cohort
Record W4403893657 · doi:10.1186/s13023-024-03370-z

Improving genetic testing pathways for transthyretin amyloidosis in France: challenges and strategies

2024· article· en· W4403893657 on OpenAlexaff
Bérénice Hebrard, Marie-Lise Babonneau, Philippe Charron, Émilie Consolino, Benjamin Dauriat, Delphine Dupin‐Deguine, Dominique Fargeaud, Agnès Farrugia, Anna‐Gaëlle Giguet‐Valard, Damien Guijarro, Jocelyn Inamo, Julien Jeanneteau, Jean‐Michaël Mazzella, Claire‐Cécile Michon, Gilles Millat, Frédéric Mouquet, Silvia Oghina, Yann Péréon, Vianney Poinsignon, Julie Pompougnac, Julie Proukhnitzky, Élise Schaefer, Franck Sturtz, Mathilde Trosdorf, Giorgia Canali, Alexandre Combes, Benoît Funalot, Thibaud Damy

Bibliographic record

VenueOrphanet Journal of Rare Diseases · 2024
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicAmyloidosis: Diagnosis, Treatment, Outcomes
Canadian institutionsHotel Dieu Hospital
FundersPfizer FrancePfizer
KeywordsGenetic testingMedicineAmyloidosisGenetic counselingDiseaseTransthyretinStakeholderStandardizationPersonalized medicineIntensive care medicineBioinformaticsGeneticsBiologyPathologyComputer sciencePolitical scienceInternal medicinePublic relations

Abstract

fetched live from OpenAlex

Transthyretin amyloidosis (ATTR) is a severe and rare disease characterized by the progressive deposition of misfolded transthyretin proteins, causing irreversible organ damage. Transthyretin amyloidosis can present as a hereditary ATTR or acquired wild-type ATTR form. Genetic testing is critical for determining a hereditary predisposition and subsequently initiating appropriate family screening. In France, strict regulations govern genetic testing that aim to protect patients and their families affected by hereditary diseases such as ATTR. However, challenges persist in establishing an effective genetic testing pathway. A multidisciplinary group of French experts convened to discuss the challenges associated with an ATTR genetic diagnosis and to propose improvement strategies. Key challenges include the lack of pathway standardization, communication gaps between healthcare professionals (HCPs) and patients, and difficulties in complying with regulatory requirements. Concerns about patient data safety and outsourced testing quality further complicate matters. Proposed strategies included the development of stakeholder mapping tools for HCPs and patients, educational programs to improve literacy on genetic testing regulations, increase disease awareness among medical geneticists and genetic counselors, and strengthening HCP-patient communication through educational materials. These initiatives aim to streamline the genetic testing pathway, enhance compliance with regulations, and ultimately provide optimal support for patients and families with ATTR.

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)0.077
metaresearch head score (Gemma)0.068
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: none
Teacher disagreement score0.077
Threshold uncertainty score0.409

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0770.068
Meta-epidemiology (narrow)0.0020.000
Meta-epidemiology (broad)0.0010.002
Bibliometrics0.0050.002
Science and technology studies0.0030.002
Scholarly communication0.0090.007
Open science0.0050.009
Research integrity0.0060.006
Insufficient payload (model declined to judge)0.0090.002

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 head0.018
GPT teacher head0.258
Teacher spread0.240 · 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 designNot applicable
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

Citations2
Published2024
Admission routes1
Has abstractyes

Explore more

Same venueOrphanet Journal of Rare DiseasesSame topicAmyloidosis: Diagnosis, Treatment, OutcomesFrench-language works237,207