Genotypic Frequencies of Mutations Associated with Alpha-1 Antitrypsin Deficiency in Unrelated Bone Marrow Donors from the Murcia Region Donor Registry in the Southeast of Spain.

AAT1 deficiency Pi system SERPINE1 gene chronic obstructive pulmonary disease

Journal

Diagnostics (Basel, Switzerland)
ISSN: 2075-4418
Titre abrégé: Diagnostics (Basel)
Pays: Switzerland
ID NLM: 101658402

Informations de publication

Date de publication:
02 Sep 2023
Historique:
received: 27 07 2023
revised: 27 08 2023
accepted: 30 08 2023
medline: 9 9 2023
pubmed: 9 9 2023
entrez: 9 9 2023
Statut: epublish

Résumé

Alpha-1 antitrypsin (AAT1) deficiency (AAT1D) is an inherited disease with an increased risk of chronic obstructive pulmonary disease (COPD), liver disease, and skin and blood vessel problems. AAT1D is caused by mutations in the SERPINE1 gene (Serine Protease Inhibitor, group A, member 1). Numerous variants of this gene, the Pi system, have been identified. The most frequent allelic variants are Pi*M, Pi*S, and Pi*Z. The development of COPD requires both a genetic predisposition and the contribution of an environmental factor, smoking being the most important. Studies on this deficiency worldwide are very scarce, and it is currently considered a rare disease because it is underdiagnosed. The aim of this study was to analyze the genotypic frequencies of mutations associated with AAT1 deficiency in unrelated bone marrow donors from the donor registry of the Region of Murcia in southeastern Spain due to the high risk of presenting with different pathologies and underdiagnosis in the population. A total of 112 DNA-healthy voluntary unrelated bone marrow donors from different parts of the Region of Murcia were analyzed retrospectively. AAT1 deficiency patient testing involved an automated biochemical screening routine. The three main variants, Pi*M, Pi*Z, and Pi*S, were analyzed in the SERPINE1 gene. Our results showed a frequency of 3.12% of the Pi*Z (K342) mutation in over 224 alleles tested in the healthy population. The frequency of Pi*S (V264) was 11.1%. The frequency of the haplotype with the most dangerous mutation, EK342 EE264, was 4.46%, and the frequency of EK342 EV264 was 1.78% in the healthy population. Frequencies of other EE342 EV264-mutated haplotypes accounted for 18.7%. As for the EE342 VV264 haplotype, 0.89% of the total healthy population presented heterozygous for the EV264 mutation and one individual presented homozygous for the VV264 mutation. In conclusion, the frequencies of Pi mutations in the healthy population of the Region of Murcia were not remarkably different from the few studies reported in Spain. The genotype and haplotype frequencies followed the usual pattern. Health authorities should be aware of this high prevalence of the Pi*S allelic variant and pathological genotypes such as Pi*MZ and Pi*SZ in the healthy population if they consider screening the smoking population.

Identifiants

pubmed: 37685383
pii: diagnostics13172845
doi: 10.3390/diagnostics13172845
pmc: PMC10486455
pii:
doi:

Types de publication

Journal Article

Langues

eng

Subventions

Organisme : Instituto de Salud Carlos III (ISCIII)
ID : P19/01194
Organisme : European Union with the European Fund of Regional Development (FEDER)
ID : "A manner to build Europe."

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Auteurs

Irene Cuenca (I)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

Carmen Botella (C)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

María Rosa Moya-Quiles (MR)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

Víctor Jimenez-Coll (V)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

José Antonio Galian (JA)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

Helios Martinez-Banaclocha (H)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

Manuel Muro-Pérez (M)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

Alfredo Minguela (A)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

Isabel Legaz (I)

Department of Legal and Forensic Medicine, Biomedical Research Institute of Murcia (IMIB), Regional Campus of International Excellence "Campus Mare Nostrum", Faculty of Medicine, University of Murcia (UMU), 30100 Murcia, Spain.

Manuel Muro (M)

Immunology Service, University Clinical Hospital "Virgen de la Arrixaca", Biomedical Research Institute of Murcia (IMIB), 30120 Murcia, Spain.

Classifications MeSH