---
title: "Bronchiectasis Severity Index Validation in Alpha-1 Antitrypsin Deficiency Patients"
id: "pubmed-42758741"
canonical_url: "https://medichelpline.com/clinical-feed/pubmed-42758741"
content_type: "clinical_feed_article"
specialty: "Critical Care"
source_name: "PubMed / NCBI"
source_url: "https://pubmed.ncbi.nlm.nih.gov/42758741/"
doi: "10.1371/journal.pone.0355956"
published_at: "2026-09-18T00:00:00.000Z"
evidence_level: "Journal Article"
license: "CC-BY-NC-4.0 / Informational Use"
---
# Bronchiectasis Severity Index Validation in Alpha-1 Antitrypsin Deficiency Patients
## Provenance & Clinical Metadata
- **Canonical URL:** https://medichelpline.com/clinical-feed/pubmed-42758741
- **Specialty:** [Critical Care](https://medichelpline.com/clinical-feed/critical-care.md)
- **Primary Source:** PubMed / NCBI
- **Source URL:** [Original Journal Publication](https://pubmed.ncbi.nlm.nih.gov/42758741/)
- **DOI:** [10.1371/journal.pone.0355956](https://doi.org/10.1371%2Fjournal.pone.0355956)
- **Published At:** 2026-09-18T00:00:00.000Z
- **Evidence Rating:** Journal Article
## Executive GIST (TL;DR)
- **Bronchiectasis** is notably found in patients with **alpha-1 antitrypsin deficiency (AATD)**. - The **Bronchiectasis Severity Index (BSI)** is a multidimensional score used to predict mortality and hospitalization. - This study seeks to validate the BSI specifically in those with AATD-related bronchiectasis. - Data was sourced from the **Birmingham AATD registry**, focusing on patients with severe AATD genotypes. - A total of **198 patients** were included, primarily of the ZZ genotype and coexisting **COPD**. - Results showed significant differences in mortality across BSI severity groups. - In **Cox regression analysis**, BSI was significantly linked to mortality. - It was also associated with decline in **gas transfer (KCO)** but not with FEV1 decline or health-related quality of life. - These findings support the validity of using the BSI in this specific cohort, suggesting prospective validation is needed in larger databases.
## Clinical Analysis & Structured Key Points
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Affiliations Expand ### Affiliations * 1 Department of Applied Health Sciences, University of Birmingham, Birmingham, United Kingdom. * 2 Department of Respiratory Medicine, University Hospitals Birmingham, Birmingham, United Kingdom. * PMID: **42758741** * DOI: [ 10.1371/journal.pone.0355956 ](https://doi.org/10.1371/journal.pone.0355956) Item in Clipboard # Validation of the Bronchiectasis Severity Index in alpha-1 antitrypsin deficiency Joshua De Soyza et al. PLoS One. 2026. Show details Display options Display options Format Abstract PubMed PMID PLoS One Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22PLoS+One%22%5Bjour%5D&sort=date&sort_order=desc) * [ Search in NLM Catalog ](https://www.ncbi.nlm.nih.gov/nlmcatalog?term=%22PLoS+One%22%5BTitle+Abbreviation%5D) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42758741/) . 2026 Sep 18;21(9):e0355956. doi: 10.1371/journal.pone.0355956. eCollection 2026. ### Authors [Joshua De Soyza](https://pubmed.ncbi.nlm.nih.gov/?term=De+Soyza+J&cauthor_id=42758741)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-1 "Department of Applied Health Sciences, University of Birmingham, Birmingham, United Kingdom.")[ 2 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-2 "Department of Respiratory Medicine, University Hospitals Birmingham, Birmingham, United Kingdom."), [Paul Ellis](https://pubmed.ncbi.nlm.nih.gov/?term=Ellis+P&cauthor_id=42758741)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-1 "Department of Applied Health Sciences, University of Birmingham, Birmingham, United Kingdom.")[ 2 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-2 "Department of Respiratory Medicine, University Hospitals Birmingham, Birmingham, United Kingdom."), [Daniella Spittle](https://pubmed.ncbi.nlm.nih.gov/?term=Spittle+D&cauthor_id=42758741)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-1 "Department of Applied Health Sciences, University of Birmingham, Birmingham, United Kingdom."), [Anita Pye](https://pubmed.ncbi.nlm.nih.gov/?term=Pye+A&cauthor_id=42758741)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-1 "Department of Applied Health Sciences, University of Birmingham, Birmingham, United Kingdom."), [Alice M Turner](https://pubmed.ncbi.nlm.nih.gov/?term=Turner+AM&cauthor_id=42758741)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-1 "Department of Applied Health Sciences, University of Birmingham, Birmingham, United Kingdom.")[ 2 ](https://pubmed.ncbi.nlm.nih.gov/42758741/#short-view-affiliation-2 "Department of Respiratory Medicine, University Hospitals Birmingham, Birmingham, United Kingdom.") ### Affiliations * 1 Department of Applied Health Sciences, University of Birmingham, Birmingham, United Kingdom. * 2 Department of Respiratory Medicine, University Hospitals Birmingham, Birmingham, United Kingdom. * PMID: **42758741** * DOI: [ 10.1371/journal.pone.0355956 ](https://doi.org/10.1371/journal.pone.0355956) Item in Clipboard Cite Display options Display options Format Abstract PubMed PMID ## Abstract Bronchiectasis is increasingly recognised in patients with alpha-1 antitrypsin deficiency (AATD), yet prognostic tools validated in general bronchiectasis populations have not been specifically evaluated in this group. The Bronchiectasis Severity Index (BSI) is a widely used multidimensional score predicting mortality, hospitalisation, and exacerbations in all-cause bronchiectasis. This study aimed to validate the BSI in a cohort of patients with AATD-bronchiectasis. Clinical data were obtained from the Birmingham AATD registry. Patients with severe AATD genotypes and CT-confirmed bronchiectasis were included, while those with non-severe genotypes or alternative causes of bronchiectasis were excluded. BSI scores were calculated using available registry data with minor adjustments reflecting the limitations of cross-sectional data collection. Associations between BSI and mortality were assessed using Kaplan-Meier survival analysis and Cox proportional hazards models adjusted for COPD, smoking status, and sex. Secondary analyses evaluated associations between BSI and lung function decline (FEV1 and KCO) and health-related quality of life (SGRQ). A total of 198 patients were included (mean age 54.3 ± 9.7 years; 53.5% male), the majority with the ZZ genotype (97.5%) and coexisting COPD (87.4%). Median BSI score was 5 (range 0-14). Mortality differed significantly across BSI severity groups (p < 0.001): estimated 1-year mortality was 1.1%, 7.4%, and 20.0% for mild, moderate, and severe disease respectively, while 4-year mortality was 5.7%, 23.7%, and 31.8%. In Cox regression analysis adjusted for COPD, smoking, and sex, BSI remained significantly associated with mortality (hazard ratio 1.13 per point increase; 95% CI 1.05-1.23; p = 0.002). BSI score was also associated with greater decline in gas transfer (KCO) (β = -0.22% per point per year; p < 0.001), but showed no significant association with FEV1 decline or SGRQ score. The Bronchiectasis Severity Index is associated with mortality risk in patients with AATD-associated bronchiectasis, supporting its use as a prognostic tool in this population. These findings provide evidence that a severity score derived from all-cause bronchiectasis cohorts can be applied to AATD, although prospective validation in larger multicentre datasets is warranted. Copyright: © 2026 De Soyza et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. [PubMed Disclaimer](https://pubmed.ncbi.nlm.nih.gov/disclaimer/) ## Conflict of interest statement The authors have read the journal’s policy and have the following competing interests: PE has received speaker fees from Chiesi, GSK, and AstraZeneca. AMT has had grants to her institution and/or honoraria from CSL Behring, Grifols, Vertex, Takeda, Chiesi, AstraZeneca, GSK, Sanofi, and Boehringer Ingelheim. JDS, DA, and AP have no conflicts of interest to declare. 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