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Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema

Up to now alpha 1-antitrypsin (AAT) augmentation therapy has been approved only for commercial use in selected adults with severe AAT deficiency-related pulmonary emphysema (i.e. PI*ZZ genotypes as well as combinations of Z, rare and null alleles expressing AAT serum concentrations <11 μmol/L). H...

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Autores principales: Blanco, Ignacio, Lara, Beatriz, de Serres, Frederick
Formato: Texto
Lenguaje:English
Publicado: BioMed Central 2011
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3094201/
https://www.ncbi.nlm.nih.gov/pubmed/21486454
http://dx.doi.org/10.1186/1750-1172-6-14
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author Blanco, Ignacio
Lara, Beatriz
de Serres, Frederick
author_facet Blanco, Ignacio
Lara, Beatriz
de Serres, Frederick
author_sort Blanco, Ignacio
collection PubMed
description Up to now alpha 1-antitrypsin (AAT) augmentation therapy has been approved only for commercial use in selected adults with severe AAT deficiency-related pulmonary emphysema (i.e. PI*ZZ genotypes as well as combinations of Z, rare and null alleles expressing AAT serum concentrations <11 μmol/L). However, the compassionate use of augmentation therapy in recent years has proven outstanding efficacy in small cohorts of patients suffering from uncommon AAT deficiency-related diseases other than pulmonary emphysema, such as fibromyalgia, systemic vasculitis, relapsing panniculitis and bronchial asthma. Moreover, a series of preclinical studies provide evidence of the efficacy of AAT augmentation therapy in several infectious diseases, diabetes mellitus and organ transplant rejection. These facts have generated an expanding number of medical applications and patents with claims for other indications of AAT besides pulmonary emphysema. The aim of the present study is to compile and analyze both clinical and histological features of the aforementioned published case studies and reports where AAT augmentation therapy was used for conditions other than pulmonary emphysema. Particularly, our research refers to ten case reports and two clinical trials on AAT augmentation therapy in patients with both AAT deficiency and, at least, one of the following diseases: fibromyalgia, vasculitis, panniculitis and bronchial asthma. In all the cases, AAT was successfully applied whereas previous maximal conventional therapies had failed. In conclusion, laboratory studies in animals and humans as well as larger clinical trials should be, thus, performed in order to determine both the strong clinical efficacy and security of AAT in the treatment of conditions other than pulmonary emphysema.
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spelling pubmed-30942012011-05-14 Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema Blanco, Ignacio Lara, Beatriz de Serres, Frederick Orphanet J Rare Dis Review Up to now alpha 1-antitrypsin (AAT) augmentation therapy has been approved only for commercial use in selected adults with severe AAT deficiency-related pulmonary emphysema (i.e. PI*ZZ genotypes as well as combinations of Z, rare and null alleles expressing AAT serum concentrations <11 μmol/L). However, the compassionate use of augmentation therapy in recent years has proven outstanding efficacy in small cohorts of patients suffering from uncommon AAT deficiency-related diseases other than pulmonary emphysema, such as fibromyalgia, systemic vasculitis, relapsing panniculitis and bronchial asthma. Moreover, a series of preclinical studies provide evidence of the efficacy of AAT augmentation therapy in several infectious diseases, diabetes mellitus and organ transplant rejection. These facts have generated an expanding number of medical applications and patents with claims for other indications of AAT besides pulmonary emphysema. The aim of the present study is to compile and analyze both clinical and histological features of the aforementioned published case studies and reports where AAT augmentation therapy was used for conditions other than pulmonary emphysema. Particularly, our research refers to ten case reports and two clinical trials on AAT augmentation therapy in patients with both AAT deficiency and, at least, one of the following diseases: fibromyalgia, vasculitis, panniculitis and bronchial asthma. In all the cases, AAT was successfully applied whereas previous maximal conventional therapies had failed. In conclusion, laboratory studies in animals and humans as well as larger clinical trials should be, thus, performed in order to determine both the strong clinical efficacy and security of AAT in the treatment of conditions other than pulmonary emphysema. BioMed Central 2011-04-12 /pmc/articles/PMC3094201/ /pubmed/21486454 http://dx.doi.org/10.1186/1750-1172-6-14 Text en Copyright ©2011 Blanco et al; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Review
Blanco, Ignacio
Lara, Beatriz
de Serres, Frederick
Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema
title Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema
title_full Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema
title_fullStr Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema
title_full_unstemmed Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema
title_short Efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema
title_sort efficacy of alpha1-antitrypsin augmentation therapy in conditions other than pulmonary emphysema
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3094201/
https://www.ncbi.nlm.nih.gov/pubmed/21486454
http://dx.doi.org/10.1186/1750-1172-6-14
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