AAT deficiency is frequently under-recognised by clinicians. Direct population-based screening has estimated that prevalence of the PI*ZZ phenotype in the US is 1 in 4455 people.[13]Stoller JK, Aboussouan LS. A review of α1-antitrypsin deficiency. Am J Respir Crit Care Med. 2012 Feb 1;185(3):246-59.
https://www.atsjournals.org/doi/full/10.1164/rccm.201108-1428CI
http://www.ncbi.nlm.nih.gov/pubmed/21960536?tool=bestpractice.com
The largest prospective screening study of newborns was performed in Sweden and reported a PI*Z phenotype prevalence of 1 in 1639.[14]Sveger T. Liver disease in alpha 1-antitrypsin deficiency detected by screening of 200,000 infants. N Engl J Med. 1976;294:1316-21.
http://www.ncbi.nlm.nih.gov/pubmed/1083485?tool=bestpractice.com
One indirect genetic survey in the US determined the frequency of the Z allele and estimated that 59,047 individuals carry the genotype.[15]de Serres FJ, Blanco I, Fernandez-Bustillo E. Genetic epidemiology of alpha-1 antitrypsin deficiency in North America and Australia/New Zealand: Australia, Canada, New Zealand and the United States of America. Clin Genet. 2003;64:382-97.
http://www.ncbi.nlm.nih.gov/pubmed/14616761?tool=bestpractice.com
Although the two greatest disease-associated alleles (S and Z) have been documented in every racial group, people with northern European ancestry carry the greatest frequencies of these alleles.[3]DeMeo DL, Silverman EK. Alpha 1-antitrypsin deficiency. 2: genetic aspects of alpha 1-antitrypsin deficiency: phenotypes and genetic modifiers of emphysema risk. Thorax. 2004;59:259-64.
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC1746953
http://www.ncbi.nlm.nih.gov/pubmed/14985567?tool=bestpractice.com
[16]de Serres FJ. Worldwide racial and ethnic distribution of alpha-1 antitrypsin deficiency. Chest. 2002;122:1818-29.
http://www.ncbi.nlm.nih.gov/pubmed/12426287?tool=bestpractice.com
Z allele frequency is highest (20-40 per 1000) in northwestern Europe and decreases towards the east of the continent.[17]Blanco I, de Serres FJ, Cárcaba V, et al. Alpha-1 antitrypsin deficiency PI*Z and PI*S gene frequency distribution using on maps of the world by an inverse distance weighting (IDW) multivariate interpolation method. Hepat Mon. 2012 Oct;12(10):e7434.
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3500828
http://www.ncbi.nlm.nih.gov/pubmed/23166537?tool=bestpractice.com
The prevalence of PI*ZZ phenotype in people with COPD is 1 in 408 in northern Europe and 1 in 944 in western Europe.[18]Blanco I, Diego I, Bueno P, et al. Prevalence of α1-antitrypsin PiZZ genotypes in patients with COPD in Europe: a systematic review. Eur Respir Rev. 2020 Sep;29(157): 200014.
https://err.ersjournals.com/content/29/157/200014
http://www.ncbi.nlm.nih.gov/pubmed/32699024?tool=bestpractice.com
A self-reported allelic frequency study utilising direct-to-consumer testing investigated the frequency of S and Z alleles in almost 200,000 people. The study demonstrated an allele frequency of 15.1% for PI*S and 6.5% for PI*Z. The PI*ZZ genotype was present in 0.63% of participants, half of whom had been diagnosed with AAT deficiency.[19]Ashenhurst JR, Nhan H, Shelton JF, et al. Prevalence of alpha-1 antitrypsin deficiency, self-reported behavior change, and health care engagement among direct-to-consumer recipients of a personalized genetic risk report. Chest. 2022 Feb;161(2):373-81.
https://journal.chestnet.org/article/S0012-3692(21)04099-X/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/34656525?tool=bestpractice.com
There is limited evidence to suggest that symptomatic lung disease is more prevalent in PI*ZZ males than in PI*ZZ females. However, this result is likely to be confounded by other variables, such as smoking and occupational exposure.[20]Kueppers F, Fallat R, Larson RK. Obstructive lung disease and alpha-1 antitrypsin deficiency gene heterozygosity. Science. 1969;165:899-901.
http://www.ncbi.nlm.nih.gov/pubmed/5816326?tool=bestpractice.com
[21]Kueppers F, Black LF. Alpha-1 antitrypsin and its deficiency. Am Rev Respir Dis. 1974;110:176-194.
http://www.ncbi.nlm.nih.gov/pubmed/4212922?tool=bestpractice.com
[22]Tobin MJ, Cook PJ, Hutchison DC. Alpha-1 antitrypsin deficiency: the clinical and physiological features of pulmonary emphysema in subjects homozygous for Pi-type-Z. A survey by the British Thoracic Association. Br J Dis Chest. 1983;77:14-27.
http://www.ncbi.nlm.nih.gov/pubmed/6602621?tool=bestpractice.com
[23]Seersholm N, Kok-Jensen A, Dirksen A. Decline in FEV1 among patients with severe hereditary alpha-1 antitrypsin deficiency type Pi Z. Am J Respir Crit Care Med. 1995;152:1922-1925.
http://www.ncbi.nlm.nih.gov/pubmed/8520756?tool=bestpractice.com
The mean age at which smokers with AAT deficiency typically present with symptomatic pulmonary disease is 32 to 41 years.[24]Larsson C. Natural history and life expectancy in severe alpha 1-antitrypsin deficiency, Pi Z. Acta Med Scand. 1978;204:345-351.
http://www.ncbi.nlm.nih.gov/pubmed/309708?tool=bestpractice.com
Liver involvement may be evident in neonates, and is the second most common cause of liver transplant in children.[14]Sveger T. Liver disease in alpha 1-antitrypsin deficiency detected by screening of 200,000 infants. N Engl J Med. 1976;294:1316-21.
http://www.ncbi.nlm.nih.gov/pubmed/1083485?tool=bestpractice.com
[25]Ghishan FK, Gray GF, Greene HL. alpha 1-antitrypsin deficiency presenting with ascites and cirrhosis in the neonatal period. Gastroenterology. 1983 Aug;85(2):435-8.
http://www.ncbi.nlm.nih.gov/pubmed/6602727?tool=bestpractice.com
Mortality is high in those with severe liver disease, and death may occur in the first decade of life.[26]Sveger T, Eriksson S. The liver in adolescents with alpha 1-antitrypsin deficiency. Hepatology. 1995 Aug;22(2):514-7.
https://www.doi.org/10.1002/hep.1840220221
http://www.ncbi.nlm.nih.gov/pubmed/7635419?tool=bestpractice.com
Liver disease is also the most common manifestation in non-smoking older adults with AAT deficiency who do not manifest pulmonary symptoms.[27]Eriksson S. Alpha 1-antitrypsin deficiency: natural course and therapeutic strategies. In: Boyer J, Blum HE, Maier KP, et al, eds. Cirrhosis and its development. Falk Symposium 115. Dordrecht, Netherlands: Kluwer Academic; 2000:307-315. Liver fibrosis has been detected in 20% to 36% of asymptomatic adults with PI*ZZ AAT deficiency, while the reported prevalence of cirrhosis ranges from 2% to 43%.[6]American Thoracic Society/European Respiratory Society Statement. Standards for the diagnosis and management of individuals with alpha 1-antitrypsin deficiency. Am J Respir Crit Care Med. 2003 Oct 1;168(7):818-900.
https://www.atsjournals.org/doi/full/10.1164/rccm.168.7.818
http://www.ncbi.nlm.nih.gov/pubmed/14522813?tool=bestpractice.com
[28]Hamesch K, Mandorfer M, Pereira VM, et al. Liver Fibrosis and Metabolic Alterations in Adults With alpha-1-antitrypsin Deficiency Caused by the Pi*ZZ Mutation. Gastroenterology. 2019 Sep;157(3):705-719.e18.
https://www.doi.org/10.1053/j.gastro.2019.05.013
http://www.ncbi.nlm.nih.gov/pubmed/31121167?tool=bestpractice.com
[29]Clark VC, Marek G, Liu C, et al. Clinical and histologic features of adults with alpha-1 antitrypsin deficiency in a non-cirrhotic cohort. J Hepatol. 2018 Dec;69(6):1357-1364.
https://www.doi.org/10.1016/j.jhep.2018.08.005
http://www.ncbi.nlm.nih.gov/pubmed/30138687?tool=bestpractice.com