Paulo Henrique Ramos Feitosa, Maria Vera Cruz de Oliveira Castellano, Claudia Henrique da Costa, Amanda da Rocha Oliveira Cardoso, Luiz Fernando Ferreira Pereira, Frederico Leon Arrabal Fernandes, Fábio Marcelo Costa, Manuela Brisot Felisbino, Alina Faria França de Oliveira, Jose R Jardim, Marc Miravitlles
{"title":"α -1抗胰蛋白酶缺乏症的诊断和治疗建议。","authors":"Paulo Henrique Ramos Feitosa, Maria Vera Cruz de Oliveira Castellano, Claudia Henrique da Costa, Amanda da Rocha Oliveira Cardoso, Luiz Fernando Ferreira Pereira, Frederico Leon Arrabal Fernandes, Fábio Marcelo Costa, Manuela Brisot Felisbino, Alina Faria França de Oliveira, Jose R Jardim, Marc Miravitlles","doi":"10.36416/1806-3756/e20240235","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":14845,"journal":{"name":"Jornal Brasileiro De Pneumologia","volume":"50 5","pages":"e20240235"},"PeriodicalIF":2.9000,"publicationDate":"2024-12-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11601085/pdf/","citationCount":"0","resultStr":"{\"title\":\"Recommendations for the diagnosis and treatment of alpha-1 antitrypsin deficiency.\",\"authors\":\"Paulo Henrique Ramos Feitosa, Maria Vera Cruz de Oliveira Castellano, Claudia Henrique da Costa, Amanda da Rocha Oliveira Cardoso, Luiz Fernando Ferreira Pereira, Frederico Leon Arrabal Fernandes, Fábio Marcelo Costa, Manuela Brisot Felisbino, Alina Faria França de Oliveira, Jose R Jardim, Marc Miravitlles\",\"doi\":\"10.36416/1806-3756/e20240235\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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. 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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.
期刊介绍:
The Brazilian Journal of Pulmonology publishes scientific articles that contribute to the improvement of knowledge in the field of the lung diseases and related areas.