Recommendations for the diagnosis and treatment of alpha-1 antitrypsin deficiency

Alpha-1 antitrypsin deficiency (AATD) is a relatively rare genetic disorder, inherited in an autosomal codominant manner, that results in reduced serum AAT concentrations, with a consequent reduction in antielastase activity in the lungs, as well as an increased risk of diseases such as pulmonary emphysema, liver cirrhosis, and necrotizing panniculitis. It results from different mutations in the SERPINA1 gene, leading to changes in the AAT glycoprotein, which can alter its concentration, conformation, and function. Unfortunately, underdiagnosis is quite common; it is possible that only 10% of cases are diagnosed. The most common deficiency is in the Z variant, and it is estimated that more than 3 million people worldwide have combinations of alleles associated with severe AATD. Serum AAT concentrations should be determined, and allelic variants should be identified by phenotyping or genotyping. Monitoring lung function, especially through spirometry, is essential, because it provides information on the progression of the disease. Although pulmonary densitometry appears to be the most sensitive measure of emphysema progression, it should not be used in routine clinical practice to monitor patients. In general, the treatment is similar to that indicated for patients with COPD not caused by AATD. Exogenous administration of purified human serum-derived AAT is the only specific treatment approved for AATD in nonsmoking patients with severe deficiency (serum AAT concentration of < 57 mg/dL or < 11 µM), with evidence of functional loss above the physiological level.

Authors

Institutions

Publication Details

Journal
Jornal Brasileiro de Pneumologia
Published
2024-11-21
DOI
https://doi.org/10.36416/1806-3756/e20240235
Citations
2
Primary Topic
Protease and Inhibitor Mechanisms
Type
article
Field-Weighted Citation Impact
0.57
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Recommendations for the diagnosis and treatment of alpha-1 antitrypsin deficiency

Luiz Fernando Ferreira Pereira, Fábio Marcelo Costa, Manuela Brisot Felisbino, Frederico Leon Arrabal Fernandes et al.
2 citations
Jornal Brasileiro de Pneumologia
Protease and Inhibitor Mechanisms
0.57
article

Recommendations for the diagnosis and treatment of alpha-1 antitrypsin deficiency

Luiz Fernando Ferreira Pereira, Fábio Marcelo Costa, Manuela Brisot Felisbino, Frederico Leon Arrabal Fernandes, A. Oliveira, Maria Vera Cruz de Oliveira Castellano, Marc Miravitlles, José Roberto Jardim, Cláudia Costa, Amanda da Rocha Oliveira Cardoso, Paulo Henrique Ramos Feitosa
article en
2 citations

Abstract

Alpha-1 antitrypsin deficiency (AATD) is a relatively rare genetic disorder, inherited in an autosomal codominant manner, that results in reduced serum AAT concentrations, with a consequent reduction in antielastase activity in the lungs, as well as an increased risk of diseases such as pulmonary emphysema, liver cirrhosis, and necrotizing panniculitis. It results from different mutations in the SERPINA1 gene, leading to changes in the AAT glycoprotein, which can alter its concentration, conformation, and function. Unfortunately, underdiagnosis is quite common; it is possible that only 10% of cases are diagnosed. The most common deficiency is in the Z variant, and it is estimated that more than 3 million people worldwide have combinations of alleles associated with severe AATD. Serum AAT concentrations should be determined, and allelic variants should be identified by phenotyping or genotyping. Monitoring lung function, especially through spirometry, is essential, because it provides information on the progression of the disease. Although pulmonary densitometry appears to be the most sensitive measure of emphysema progression, it should not be used in routine clinical practice to monitor patients. In general, the treatment is similar to that indicated for patients with COPD not caused by AATD. Exogenous administration of purified human serum-derived AAT is the only specific treatment approved for AATD in nonsmoking patients with severe deficiency (serum AAT concentration of < 57 mg/dL or < 11 µM), with evidence of functional loss above the physiological level.

Jornal Brasileiro de PneumologiaVol. 50(5)
Hospital de Base (BR), Universidade do Estado do Rio de Janeiro (BR), Universidade Federal de Santa Catarina (BR), Secretaria de Educação de Pernambuco (BR), Vall d'Hebron Institut de Recerca (ES), Polydoro Ernani de São Thiago University Hospital (BR), Hospital das Clínicas da Universidade Federal de Minas Gerais (BR), Hospital de Clínicas Universidade Federal do Paraná (BR), Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo (BR), Hospital do Servidor Público Estadual (BR), Universidade Federal de Goiás (BR), Universidade Federal de São Paulo (BR)
Good health and well-being
Openalex Percentile: Top 30%
Protease and Inhibitor Mechanisms
0.57
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.