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3. Triple Therapy and Clinical Control in B+ COPD Patients: A Pragmatic, Prospective, Randomized Trial

Author

Agusti, Alvar, Lopez-Campos, José Luis, Miravitlles, Marc, Soler-Cataluña, Juan Jose, Marin, Jose Maria, Cosio, Borja G., Alcázar-Navarrete, Bernardino, Echave-Sustaeta, Jose Maria, Casanova, Ciro, Peces-Barba, German, de-Torres, Juan Pablo, Fernandez-Villar, Alberto, Ancochea, Julio, Villar-Alvarez, Felipe, Roman-Rodriguez, Miguel, Molina, Jesus, Garcia-Rivero, Juan Luis, Gonzalez, Cruz, Sobradillo, Patricia, Faner, Rosa, Peña, Carolina, Sharma, Raj, Izquierdo, José Luis, and Celli, Bartolome R.

Published
2024
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6. Effect of different types of supervised exercise programs on cardiorespiratory and muscular fitness, pain, fatigue, mental health and inflammatory and oxidative stress biomarkers in older patients with post-COVID-19 sequelae “EJerSA-COVID-19”: a randomized controlled trial

Author

Pleguezuelos, Eulogio, Sánchez-Nuño, Sergio, Del Carmen, Amin, Serra-Payá, Noemí, Moreno, Eva, Molina-Raya, Lorena, Robleda, Gemma, Benet, Marta, Santos-Ruiz, Susana, Garrido, Ainoa Biurrun, Jerez-Molina, Carmen, Miravitlles, Marc, Serra-Prat, Mateu, Viñals, Xavier, Farrés, Montserrat Girabent, Carbonell, Teresa, and Garnacho-Castaño, Manuel V.

Published
2023
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9. COVID-19’s impact on care practice for alpha-1-antitrypsin deficiency patients

Author

Calle Rubio, Myriam, López-Campos, José Luis, Miravitlles, Marc, Michel de la Rosa, Francisco Javier, Hernández Pérez, José María, Montero Martínez, Carmen, Montoro Ronsano, José Bruno, Casas Maldonado, Francisco, Rodríguez Hermosa, Juan Luis, Tabernero Huguet, Eva María, Martínez Sesmero, José Manuel, Martínez Rivera, Carlos, Callejas González, Francisco Javier, and Torres Durán, María

Published
2023
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10. Correction: Clinical and functional characteristics of individuals with alpha-1 antitrypsin deficiency: EARCO international registry

Author

Miravitlles, Marc, Turner, Alice M., Torres-Duran, María, Tanash, Hanan, Rodríguez-García, Carlota, López-Campos, José Luis, Chlumsky, Jan, Guimaraes, Catarina, Rodríguez-Hermosa, Juan Luis, Corsico, Angelo, Martinez-González, Cristina, Hernández-Pérez, José María, Bustamante, Ana, Parr, David G., Casas-Maldonado, Francisco, Hecimovic, Ana, Janssens, Wim, Lara, Beatriz, Barrecheguren, Miriam, González, Cruz, Stolk, Jan, Esquinas, Cristina, and Clarenbach, Christian F.

Published
2023
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11. Alpha-1 Antitrypsin Augmentation and the Liver Phenotype of Adults With Alpha-1 Antitrypsin Deficiency (Genotype Pi∗ZZ)

Author

Fromme, Malin, Hamesch, Karim, Schneider, Carolin V., Mandorfer, Mattias, Pons, Monica, Thorhauge, Katrine H., Pereira, Vitor, Sperl, Jan, Frankova, Sona, Reichert, Matthias C., Benini, Federica, Burbaum, Barbara, Kleinjans, Moritz, Amzou, Samira, Rademacher, Laura, Bewersdorf, Lisa, Verbeek, Jef, Nevens, Frederik, Genesca, Joan, Miravitlles, Marc, Nuñez, Alexa, Schaefer, Benedikt, Zoller, Heinz, Janciauskiene, Sabina, Waern, Johan, Oliveira, António, Maia, Luís, Simões, Carolina, Mahadeva, Ravi, Fraughen, Daniel D., Trauner, Michael, Krag, Aleksander, Lammert, Frank, Bals, Robert, Gaisa, Nadine T., Aigner, Elmar, Griffiths, William J., Denk, Helmut, Teumer, Alexander, McElvaney, Noel G., Turner, Alice M., Trautwein, Christian, and Strnad, Pavel

Published
2024
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12. Sex-Differences in Alpha-1 Antitrypsin Deficiency: Data From the EARCO Registry

Author

Ersöz, Hilal, Torres-Durán, María, Turner, Alice M., Tanash, Hanan, García, Carlota Rodríguez, Corsico, Angelo Guido, López-Campos, José Luis, Miravitlles, Marc, Clarenbach, Christian F., Chapman, Kenneth R., Pérez, Jose Mª Hernández, Guimarães, Catarina, Bartošovská, Eva, Greulich, Timm, Barrecheguren, Miriam, Koczulla, Andreas Rembert, Höger, Philipp, Rivera, Arturo Olivares, Herth, Felix, and Trudzinski, Franziska C.

Published
2024
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18. Tobacco Patterns and Risk of Chronic Obstructive Pulmonary Disease: Results From a Cross-Sectional Study

Author

Rey-Brandariz, Julia, Pérez-Ríos, Mónica, Ahluwalia, Jasjit S., Beheshtian, Kiana, Fernández-Villar, Alberto, Represas-Represas, Cristina, Piñeiro, María, Alfageme, Inmaculada, Ancochea, Julio, Soriano, Joan B., Casanova, Ciro, Cosío, Borja G., García-Río, Francisco, Miravitlles, Marc, de Lucas, Pilar, Rodríguez González-Moro, José Miguel, Soler-Cataluña, Juan José, and Ruano-Ravina, Alberto

Published
2023
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19. Alpha-1 antitrypsin (AAT) augmentation therapy in individuals with the PI*MZ genotype: a pro/con debate on a working hypothesis.

Author

Barjaktarevic, Igor and Miravitlles, Marc

Subjects
Alpha-1 antitrypsin deficiency, Chronic obstructive pulmonary disease, Genotype, PI*MZ, Pulmonary disease, Genotype, Humans, Practice Guidelines as Topic, Pulmonary Disease, Chronic Obstructive, alpha 1-Antitrypsin, alpha 1-Antitrypsin Deficiency
Abstract

Alpha-1 antitrypsin deficiency (AATD) is a significantly under-diagnosed genetic condition caused by reduced levels and/or functionality of alpha-1 antitrypsin (AAT), predisposing individuals to lung, liver or other systemic diseases. The management of individuals with the PI*MZ genotype, characterized by mild or moderate AAT deficiency, is less clear than of those with the most common severe deficiency genotype (PI*ZZ). Recent genetic data suggest that the PI*MZ genotype may be significantly more prevalent than currently thought. The only specific treatment for lung disease associated with severe AATD is the intravenous infusion of AAT augmentation therapy, which has been shown to slow disease progression in PI*ZZ individuals. There is no specific evidence for the clinical benefit of AAT therapy in PI*MZ individuals, and the risk of emphysema development in this group remains controversial. As such, current guidelines do not support the use of AAT augmentation in PI*MZ individuals. Here, we discuss the limited data on the PI*MZ genotype and offer pro and con perspectives on pursuing an AAT-specific therapeutic strategy in PI*MZ individuals with lung disease. Ultimately, further research to demonstrate the safety, risk/benefit balance and efficacy of AAT therapy in PI*MZ individuals is needed.

Published
2021

24. Leveraging CT Imaging to Detect COPD and Concomitant Chronic Diseases

Author

Labaki, Wassim W., primary, Agusti, Alvar, additional, Bhatt, Surya P, additional, Bodduluri, Sandeep, additional, Criner, Gerard J., additional, Fabbri, Leonardo M, additional, Halpin, David M.G., additional, Lynch, David A., additional, Mannino, David M., additional, Miravitlles, Marc, additional, Papi, Alberto, additional, Sin, Don D, additional, Washko, George R, additional, Kazerooni, Ella A., additional, and Han, MeiLan K., additional

Published
2024
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25. WED-135-YI Impact of metabolic alterations on liver phenotype of patients with homozygous alpha-1 antitrypsin deficiency (Pi*ZZ)

Author

Schrader, Christina, primary, Fromme, Malin, additional, Payancé, Audrey, additional, Mandorfer, Mattias, additional, Thorhauge, Katrine, additional, Pons, Monica, additional, Miravitlles, Marc, additional, Stolk, Jan, additional, van Hoek, Bart, additional, Stirnimann, Guido, additional, Fraňková, Soňa, additional, Sperl, Jan, additional, Kremer, Andreas E., additional, Balcar, Lorenz, additional, Schaefer, Benedikt, additional, Chorostowska-Wynimko, Joanna, additional, Trautwein, Christian, additional, Zoller, Heinz, additional, Trauner, Michael, additional, Genesca, Joan, additional, Griffiths, William J.H., additional, Clark, Virginia C., additional, Krag, Aleksander, additional, McElvaney, Noel G., additional, Turner, Alice, additional, and Strnad, Pavel, additional

Published
2024
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26. TOP-141 Longitudinal assessment of individuals with homozygous alpha-1 antitrypsin deficiency (Pi*ZZ genotype) provides evidence for clinical patient management

Author

Fromme, Malin, primary, Payancé, Audrey, additional, Mandorfer, Mattias, additional, Thorhauge, Katrine, additional, Pons, Monica, additional, Miravitlles, Marc, additional, Stolk, Jan, additional, van Hoek, Bart, additional, Stirnimann, Guido, additional, Fraňková, Soňa, additional, Sperl, Jan, additional, Kremer, Andreas E., additional, Schrader, Christina, additional, Balcar, Lorenz, additional, Schaefer, Benedikt, additional, Chorostowska-Wynimko, Joanna, additional, Ryan, John, additional, Trautwein, Christian, additional, Zoller, Heinz, additional, Trauner, Michael, additional, Genesca, Joan, additional, Griffiths, William J.H., additional, Clark, Virginia C., additional, Krag, Aleksander, additional, Turner, Alice, additional, McElvaney, Noel G., additional, and Strnad, Pavel, additional

Published
2024
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29. Concomitant inhaled corticosteroid use and the risk of pneumonia in COPD: a matched-subgroup post hoc analysis of the UPLIFT® trial

Author

Tashkin, Donald P, Miravitlles, Marc, Celli, Bartolomé R, Metzdorf, Norbert, Mueller, Achim, Halpin, David MG, and Anzueto, Antonio

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Pneumonia & Influenza, Pneumonia, Lung, Clinical Research, Respiratory, Administration, Inhalation, Adrenal Cortex Hormones, Aged, Bronchodilator Agents, Double-Blind Method, Female, Fluticasone, Humans, Male, Middle Aged, Pulmonary Disease, Chronic Obstructive, Research Design, Risk Factors, Tiotropium Bromide, COPD, Inhaled corticosteroids, Fluticasone propionate, UPLIFT, UPLIFT®, Cardiorespiratory Medicine and Haematology, Respiratory System, Cardiovascular medicine and haematology, Clinical sciences
Abstract

BackgroundUse of inhaled corticosteroids (ICS) increases the risk of pneumonia in chronic obstructive pulmonary disease (COPD), but the magnitude of risk with different ICS remains unclear.MethodsA post hoc analysis of the 4-year UPLIFT® trial to assess whether pneumonia risk differed by type of ICS (fluticasone propionate [FP], other ICS, or no ICS) in permanent users (defined by use until end of study) or in users at baseline (sensitivity analysis).ResultsFor the permanent-users analysis, 825 patients receiving FP throughout the trial, 825 patients receiving other ICS and 825 patients not receiving ICS were matched on relevant baseline features 1:1:1. A significantly greater risk of pneumonia was observed for FP versus no ICS: the hazard ratio (HR) for risk of pneumonia was 1.33 (95% confidence interval [CI] 1.00, 1.75; p = 0.046) and the rate ratio (RR) was 1.58 (95% CI 1.05, 2.37; p = 0.028). A greater risk was also found for FP versus other ICS: HR 1.28 (95% CI 0.97, 1.68; p = 0.078) and RR 1.48 (95% CI 1.00, 2.19; p = 0.049). A higher proportion of patients on FP were hospitalized with pneumonia (7.9%) versus other ICS (6.7%) or no ICS (5.9%). Whilst other ICS use was associated with the highest number of fatal pneumonia events, the total number of fatal pneumonia incidents was low. A similar pattern was observed in the sensitivity analyses, which included 4002 patients on different treatments at baseline (FP, other ICS, and no ICS) and considered potential switches during the study.ConclusionThe results support existing evidence of an increased pneumonia risk with FP use compared with other ICS and no ICS use in patients with COPD. Healthcare professionals should evaluate the risk-benefit ratio of using ICS when making treatment decisions with their patients.Trial registrationPost hoc analysis of UPLIFT®. ClinicalTrials.gov number: NCT00144339 . Retrospectively registered September 2, 2005.

Published
2018

39. Clinical and functional characteristics of individuals with alpha-1 antitrypsin deficiency: EARCO international registry

Author

Miravitlles, Marc, Turner, Alice M., Torres-Duran, María, Tanash, Hanan, Rodríguez-García, Carlota, López-Campos, José Luis, Chlumsky, Jan, Guimaraes, Catarina, Rodríguez-Hermosa, Juan Luis, Corsico, Angelo, Martinez-González, Cristina, Hernández-Pérez, José María, Bustamante, Ana, Parr, David G., Casas-Maldonado, Francisco, Hecimovic, Ana, Janssens, Wim, Lara, Beatriz, Barrecheguren, Miriam, González, Cruz, Stolk, Jan, Esquinas, Cristina, and Clarenbach, Christian F.

Published
2022
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45. Effectiveness and Safety of Inhaled Antibiotics in Patients With Chronic Obstructive Pulmonary Disease. A Multicentre Observational Study

Author

Rolon, Annie Navarro, Wang, Xuejie, Tapia, Alicia Marín, Rubio, Myriam Calle, Asensio, María Jesús Linares, Orbis, Iria Pérez, Olondris, Pilar Martínez, Sanz, Ascensión Hernando, Gafas, Alicia de Pablos, Royo, Margarita Marín, Peris, Selene Cuenca, Fuertes, Julia Amaranta García, Olveira, Casilda, Ramos, Guillermo Bentabol, Bou, Lirios Sacristán, Moreno, Rosa María Girón, Arguedas, Sandra Marín, Zabaleta, Raúl Moreno, Fernández, Sarai Quirós, Sarasate, Mikel, Arranz, María Victoria Leal, Pozas, Gema Castaño de las, Ávila, Nuria Bruguera, Diago, Carlos Antonio Amado, Viteri, Soledad Alonso, Cancelo, María Isabel Ramos, Rivera, Carolina Gotera, Díez, Javier de Miguel, Muñoz, Gemma Sánchez, Zapatero, Esperanza Martín, Celis, Sandra Ros, Navarro, Silvia Merlos, García, Rut Ayerbe, De la Rosa Carrillo, David, Martínez-García, Miguel Ángel, Barreiro, Esther, Tabernero Huguet, Eva, Costa Sola, Roser, García-Clemente, Marta María, Celorrio Jiménez, Nuria, Rodríguez Pons, Laura, Calero Acuña, Carmen, Rodríguez Hermosa, Juan Luís, Golpe, Rafael, Dacal Quintas, Raquel, Sánchez-Cuéllar, Silvia, Torres Arroyo, Irene, Blanco Aparicio, Marina, Almadana Pacheco, Virginia, and Miravitlles, Marc

Published
2022
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47. Assessment and monitoring of lung disease in patients with severe alpha 1 antitrypsin deficiency: a european delphi consensus of the EARCO group.

Author

Miravitlles, Marc, Turner, Alice M., Sucena, Maria, Mornex, Jean-François, Greulich, Timm, Wencker, Marion, and McElvaney, N. Gerard

Subjects
*ALPHA 1-antitrypsin, *DELPHI method, *COMPUTED tomography, *DISEASE management, *PHYSICIANS, *ALPHA 1-antitrypsin deficiency
Abstract

Background: Currently, there is conflicting information and guidance on the effective management of Alpha 1 Antitrypsin Deficiency (AATD). Establishing a consensus of assessment and disease management specific to AATD is important for achieving a standardized treatment pathway and for improving patient outcomes. Here, we aim to utilize the Delphi method to establish a European consensus for the assessment and management of patients with severe AATD. Methods: Two rounds of a Delphi survey were completed online by members of the European Alpha-1 Research Collaboration (EARCO). Respondents were asked to indicate their agreement with proposed statements for patients with no respiratory symptoms, stable respiratory disease, and worsening respiratory disease using a Likert scale of 1–7. Levels of agreement between respondents were calculated using a weighted average. Results: Round 1 of the Delphi survey was sent to 103 members of EARCO and 38/103 (36.9%) pulmonologists from across 15 countries completed all 109 questions. Round 2 was sent to all who completed Round 1 and 36/38 (94.7%) completed all 79 questions. Responses regarding spirometry, body plethysmography, high-resolution computed tomography, and the initiation of augmentation therapy showed little variability among physicians, but there was discordance among other aspects, such as the use of low-dose computed tomography in both a research setting and routine clinical care. Conclusions: These results provide expert opinions for the assessment and monitoring of patients with severe AATD, which could be used to provide updated recommendations and standardized treatment pathways for patients across Europe. [ABSTRACT FROM AUTHOR]

Published
2024
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48. Personalised indication of augmentation therapy for emphysema associated with severe alpha-1 antitrypsin deficiency: a case series.

Author

Aljama, Cristina, Martin, Teresa, Granados, Galo, Miravitlles, Marc, and Barrecheguren, Miriam

Subjects
TRAVEL restrictions, INTRAVENOUS therapy, TRYPSIN inhibitors, LUNG diseases, ALPHA 1-antitrypsin deficiency, BLOOD donors
Abstract

Severe alpha-1 antitrypsin deficiency (AATD) is associated with an increased risk of emphysema. However, the clinical manifestations are very heterogeneous, and an individual prognosis is very difficult to establish. Intravenous augmentation therapy with alpha-1 antitrypsin (AAT) from pooled blood donors is the only specific treatment available, but it requires weekly or biweekly administration for life. Several guidelines provide the indication criteria for the initiation of AAT augmentation therapy. However, in clinical practice, there are situations in which the decision as to when to start treatment becomes uncertain and some studies have shown great variability in the indication of this treatment even among specialists. The usual dilemma is between initiating augmentation therapy in individuals who may not develop significant lung disease or in whom disease will not progress or delaying it in patients who may otherwise rapidly and irreversibly progress. We illustrate this dilemma with five clinical cases: from the case of a patient with normal lung function who requests initiation of therapy to a moderately stable patient without augmentation or a mild patient who, after several years of remaining stable without treatment, deterioration in lung function initiated and, consequently, augmentation therapy was begun. All the nuances associated with the indication of augmentation justify a personalised approach and the decision about initiating augmentation therapy must be made after careful consideration of the pros and cons with the patient in reference centres with experience in treatment. These reference centres can work in collaboration with local hospitals where patients can be closely followed and augmentation therapy can be administered to avoid unnecessary travelling, making periodical administrations more comfortable for the patient. [ABSTRACT FROM AUTHOR]

Published
2024
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49. Leveraging Computed Tomography Imaging to Detect Chronic Obstructive Pulmonary Disease and Concomitant Chronic Diseases.

Author

Labaki, Wassim W., Agusti, Alvar, Bhatt, Surya P., Bodduluri, Sandeep, Criner, Gerard J., Fabbri, Leonardo M., Halpin, David M. G., Lynch, David A., Mannino, David M., Miravitlles, Marc, Papi, Alberto, Sin, Don D., Washko, George R., Kazerooni, Ella A., and Han, MeiLan K.

Subjects
CHRONIC obstructive pulmonary disease, COMPUTED tomography, DISEASE complications, BRONCHIECTASIS, INTERSTITIAL lung diseases, COMORBIDITY, OBSTRUCTIVE lung diseases
Abstract

The article addresses the global impact of chronic obstructive pulmonary disease (COPD), noting its high prevalence and the significant burden of undiagnosed cases. Topics include the limitations of current screening practices and the U.S. Preventive Services Task Force's stance against routine spirometry for asymptomatic individuals, despite evidence suggesting that at-risk populations could benefit from early detection.

Published
2024
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50. Genetic Epidemiology of Alpha-1 Antitrypsin Deficiency in Macaronesia.

Author

Blanco, Ignacio and Miravitlles, Marc

Subjects
*MEDICAL information storage & retrieval systems, *SYSTEMATIC reviews, *MEDLINE, *ALPHA 1-antitrypsin deficiency, *MEDICAL screening, *ALLELES
Abstract

Introduction: The prevalence of alpha-1 antitrypsin deficiency (AATD) in Macaronesia (i.e., Azores, Madeira, Canary Islands, and Cape Verde archipelagos) is poorly known. Our goal was to update it by selecting the most reliable available articles. Method: Literature search using MEDLINE, Embase (via Ovid), and Google Scholar, until December 2023, for studies on prevalence of AATD in the general population and in screenings, published in peer-reviewed journals. Results: Three studies carried out in the general population of Madeira, La Palma, and Cape Verde, and three screenings carried out in La Palma (2) and Gran Canaria (1) were selected. The frequencies of PI*S in the general population showed an ascending gradient, from South to North, with values (per thousand) of 35 in Cape Verde, 82 in La Palma, and 180 in Madeira. The PI*Z frequencies showed this same gradient, with values of 2 × 1,000 in Cape Verde, 21 in La Palma, and 25 in Madeira. Screenings detected high percentages of defective alleles, including several rare and null alleles, some unique to these islands. Conclusion: The frequencies of PI*S and PI*Z in Madeira are comparable to the highest in the world. Those of the Canary Islands are similar to those of the peninsular population of Spain, and contrast with the low rates of Cape Verde. Screenings detected high numbers of deficient alleles. These results support the systematic investigation of AATD in clinically suspected patients and in relatives of index cases, to reduce underdiagnosis and apply early preventive and therapeutic measures in those affected. [ABSTRACT FROM AUTHOR]

Published
2024
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