α -1抗胰蛋白酶缺乏症的诊断和治疗建议。

IF 2.9 4区 医学 Q2 RESPIRATORY SYSTEM
Jornal Brasileiro De Pneumologia Pub Date : 2024-12-06 eCollection Date: 2024-01-01 DOI:10.36416/1806-3756/e20240235
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. 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\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Jornal Brasileiro De Pneumologia\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.36416/1806-3756/e20240235\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q2\",\"JCRName\":\"RESPIRATORY SYSTEM\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Jornal Brasileiro De Pneumologia","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.36416/1806-3756/e20240235","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/1/1 0:00:00","PubModel":"eCollection","JCR":"Q2","JCRName":"RESPIRATORY SYSTEM","Score":null,"Total":0}
引用次数: 0

摘要

α -1抗胰蛋白酶缺乏症(AATD)是一种相对罕见的遗传性疾病,以常染色体共显性方式遗传,导致血清AAT浓度降低,随之而来的是肺部抗弹性酶活性降低,以及肺气肿、肝硬化和坏死性pannicitis等疾病的风险增加。它是由SERPINA1基因的不同突变引起的,导致AAT糖蛋白的变化,从而改变其浓度、构象和功能。不幸的是,诊断不足很常见;可能只有10%的病例被诊断出来。最常见的缺陷是Z变异,据估计,全世界有超过300万人具有与严重AATD相关的等位基因组合。应测定血清AAT浓度,并通过表型或基因分型确定等位变异。监测肺功能,特别是通过肺活量测定法,是必不可少的,因为它提供了疾病进展的信息。尽管肺密度测量似乎是肺气肿进展的最敏感的测量,但它不应用于常规临床实践中监测患者。一般来说,治疗方法与非AATD引起的COPD患者的治疗方法相似。外源性给药纯化的人血清源性AAT是唯一被批准用于严重缺乏AAT(血清AAT浓度< 57 mg/dL或< 11µM)且有证据表明功能丧失高于生理水平的非吸烟患者的AATD的特异性治疗。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
Jornal Brasileiro De Pneumologia
Jornal Brasileiro De Pneumologia RESPIRATORY SYSTEM-
CiteScore
3.50
自引率
14.80%
发文量
118
审稿时长
20 weeks
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.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:604180095
Book学术官方微信