Feitosa Paulo Henrique Ramos, Castellano Maria Vera Cruz de Oliveira, Costa Claudia Henrique da, Cardoso Amanda da Rocha Oliveira, Pereira Luiz Fernando Ferreira, Fernandes Frederico Leon Arrabal, Costa Fábio Marcelo, Felisbino Manuela Brisot, Oliveira Alina Faria França de, Jardim Jose R, Miravitlles Marc
. Hospital Regional da Asa Norte, Brasília (DF) Brasil.
. Hospital do Servidor Público Estadual de São Paulo - IAMSPE - São Paulo (SP) Brasil.
J Bras Pneumol. 2024 Dec 6;50(5):e20240235. doi: 10.36416/1806-3756/e20240235. eCollection 2024.
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.
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,会导致血清α-1抗胰蛋白酶(AAT)浓度降低,进而使肺部抗弹性蛋白酶活性降低,同时增加患肺气肿、肝硬化和坏死性脂膜炎等疾病的风险。它是由SERPINA1基因的不同突变引起的,导致AAT糖蛋白发生变化,从而改变其浓度、构象和功能。不幸的是,漏诊情况相当普遍;可能只有10%的病例得到诊断。最常见的缺陷是Z变体,据估计,全球有超过300万人携带与严重AATD相关的等位基因组合。应测定血清AAT浓度,并通过表型分析或基因分型鉴定等位基因变体。监测肺功能,尤其是通过肺活量测定法,至关重要,因为它能提供疾病进展的信息。尽管肺密度测定似乎是肺气肿进展最敏感的指标,但在常规临床实践中不应将其用于监测患者。一般来说,治疗方法与非AATD引起的慢性阻塞性肺疾病(COPD)患者的治疗方法相似。对于严重缺乏(血清AAT浓度<57mg/dL或<11µM)且有生理水平以上功能丧失证据的非吸烟AATD患者,外源性给予纯化的人血清源性AAT是唯一被批准的特异性治疗方法。