MétaCan
Menu
Back to cohort
Record W4413397744 · doi:10.1183/23120541.00491-2025

Characteristics of bronchiectasis in patients with different genotypes of severe α <sub>1</sub> -antitrypsin deficiency from the EARCO registry

2025· article· en· W4413397744 on OpenAlexaff
Francesca Mandurino Mirizzi, Cristina Aljama, Pierachille Santus, Marco Mantero, Maja Omčikus, María Torres‐Durán, Alice Turner, Hanan Tanash, Carlota Rodríguez‐García, Jens‐Ulrik Stæhr Jensen, Angelo Guido Corsico, José Luís López-Campos, Kenneth R. Chapman, Christian F. Clarenbach, C. Guimarães, Eva Bartošovská, José María Hernández Pérez, Marc Miravitlles, Cristina Esquinas, Míriam Barrecheguren

Bibliographic record

VenueERJ Open Research · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicProtease and Inhibitor Mechanisms
Canadian institutionsToronto Western HospitalUniversity Health Network
FundersMereo BioPharmaGrifolsEuropean Respiratory SocietySanofiTakeda Medical Research FoundationCSL BehringGlaxoSmithKlineRegeneron PharmaceuticalsTeva Pharmaceutical IndustriesAstraZeneca
KeywordsMedicineBronchiectasisGenotypeInternal medicinePediatricsLungGeneticsGene

Abstract

fetched live from OpenAlex

Background α-1 antitrypsin deficiency (AATD) is a rare genetic disorder caused by mutations in the SERPINA1 gene and associated with reduced levels of α-1 antitrypsin (AAT). It predisposes individuals to pulmonary diseases, including bronchiectasis, through protease–antiprotease imbalance and immune dysregulation. While the Pi*ZZ genotype has been extensively studied, the prevalence and characteristics of bronchiectasis in other genotypes remain unclear. Methods This cross-sectional study analysed data from the European α-1 Research Collaboration (EARCO) registry, focusing on individuals with bronchiectasis on computed tomography (CT). Participants were stratified by AATD genotypes (Pi*ZZ , Pi*SZ, Pi*SS and rare variants) and data were compared. Disease severity was evaluated using FACED (forced expiratory volume in 1 s (FEV 1 ), age, chronic colonisation, extension and dyspnoea) score and bronchiectasis severity index (BSI) scores. Results 349 patients had bronchiectasis on a CT scan, of whom 70.5% had Pi*ZZ, 18.6% had Pi*SZ, 4.3% had Pi*SS and 6.6% had rare variants. Lower lobe involvement was predominant across genotypes, whereas Pi*SS exhibited distinct upper lobe patterns and Pi*SZ showed more frequent middle lobe involvement. People with rare genotypes and Pi*ZZ had worse lung function (FEV 1 % of 65.3% and 71.4%, respectively) and higher disease severity scores. Emphysema co-occurrence was most frequent in Pi*ZZ (60.6%). No significant differences were observed in sputum microbiology or systemic inflammatory markers, except for lower platelet counts in Pi*ZZ subjects. Conclusion Bronchiectasis in AATD is not limited to the Pi*ZZ genotype, with significant phenotypic variability across genotypes. Lower lobe involvement and mild disease predominate; however, severe forms are more frequent in rare genotypes and Pi*ZZ. These findings underscore the importance of systematic screening and genotype-specific management to improve patient outcomes.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation 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 score0.138
Threshold uncertainty score0.286

Codex and Gemma teacher scores by category

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

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 head0.018
GPT teacher head0.289
Teacher spread0.271 · 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 teacher head, 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

Citations1
Published2025
Admission routes1
Has abstractyes

Explore more

Same venueERJ Open ResearchSame topicProtease and Inhibitor MechanismsFrench-language works237,207