Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency

2023 Impact Factor; 11.4. In Press - Pre-proof. “This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo add...

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Main Authors: Kahn, Adrián, Luque, Gabriela, Cuestas, Eduardo, Basquiera, Ana, Ricci, Brenda, Schmitz-Abe, Klaus, Charbonnier, Louis-Marie, Benamar, Mehdi, Motrich, Rubén Darío, Chatila, Talal A., Rivero, Virginia E.
Other Authors: https://orcid.org/0000-0002-5772-1285
Format: info:eu-repo/semantics/acceptedVersion
Language:eng
Published: American Academy of Allergy 2024
Subjects:
Online Access:http://hdl.handle.net/11086/553072
https://www.sciencedirect.com/science/article/pii/S2772829324001073
https://www.jaci-global.org/article/S2772-8293(24)00107-3/fulltext
https://doi.org/10.1016/j.jacig.2024.100311
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author Kahn, Adrián
Luque, Gabriela
Cuestas, Eduardo
Basquiera, Ana
Ricci, Brenda
Schmitz-Abe, Klaus
Charbonnier, Louis-Marie
Benamar, Mehdi
Motrich, Rubén Darío
Chatila, Talal A.
Rivero, Virginia E.
author2 https://orcid.org/0000-0002-5772-1285
author_facet https://orcid.org/0000-0002-5772-1285
Kahn, Adrián
Luque, Gabriela
Cuestas, Eduardo
Basquiera, Ana
Ricci, Brenda
Schmitz-Abe, Klaus
Charbonnier, Louis-Marie
Benamar, Mehdi
Motrich, Rubén Darío
Chatila, Talal A.
Rivero, Virginia E.
author_sort Kahn, Adrián
collection Repositorio Digital Universitario
description 2023 Impact Factor; 11.4. In Press - Pre-proof. “This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain”. Fuente: Elsevier Inc. on behalf of the American Academy of Allergy. “Este es un archivo PDF de un artículo que ha sufrido mejoras después de su aceptación, como la adición de una portada y metadatos, y formato para facilitar la lectura, pero aún no es la versión definitiva de registro. Esta versión se someterá a correcciones, composición tipográfica y revisión adicionales antes de su publicación en su forma final, pero proporcionamos esta versión para brindar visibilidad temprana del artículo. Tenga en cuenta que, durante el proceso de producción se pueden descubrir errores que podrían afectar el contenido, y todas las medidas legales Se refieren las exenciones de responsabilidad que se aplican a la revista”. Fuente: Elsevier Inc. on behalf of the American Academy of Allergy.
format info:eu-repo/semantics/acceptedVersion
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institution Universidad Nacional de Cordoba
language eng
publishDate 2024
publisher American Academy of Allergy
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spelling rdu-unc.5530722024-08-05T06:24:40Z Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency Kahn, Adrián Luque, Gabriela Cuestas, Eduardo Basquiera, Ana Ricci, Brenda Schmitz-Abe, Klaus Charbonnier, Louis-Marie Benamar, Mehdi Motrich, Rubén Darío Chatila, Talal A. Rivero, Virginia E. https://orcid.org/0000-0002-5772-1285 CVID Common variable immunodeficiency Score Mortality, Outcomes WES Whole exome sequencing B cells subpopulations T cells 2023 Impact Factor; 11.4. In Press - Pre-proof. “This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain”. Fuente: Elsevier Inc. on behalf of the American Academy of Allergy. “Este es un archivo PDF de un artículo que ha sufrido mejoras después de su aceptación, como la adición de una portada y metadatos, y formato para facilitar la lectura, pero aún no es la versión definitiva de registro. Esta versión se someterá a correcciones, composición tipográfica y revisión adicionales antes de su publicación en su forma final, pero proporcionamos esta versión para brindar visibilidad temprana del artículo. Tenga en cuenta que, durante el proceso de producción se pueden descubrir errores que podrían afectar el contenido, y todas las medidas legales Se refieren las exenciones de responsabilidad que se aplican a la revista”. Fuente: Elsevier Inc. on behalf of the American Academy of Allergy. info:eu-repo/semantics/acceptedVersion Fil: Kahn, Adrián. Hospital Privado Universitario de Córdoba. Servicio de Alergia e Inmunología Clínica; Argentina. Fil: Kahn, Adrián. Instituto Universitario de Ciencias Biomédicas de Córdoba; Argentina. Fil: Kahn, Adrián. Federation of Clinical Immunology Societies. Centro de Inmunología Clínica de Córdoba; Argentina. Fil: Luque, Gabriela. Hospital Privado Universitario de Córdoba. Servicio de Oncohematologia; Argentina. Fil: Cuestas, Eduardo. Instituto Universitario de Ciencias Biomédicas de Córdoba; Argentina. Fil: Cuestas, Eduardo. Hospital Privado Universitario de Córdoba. Servicio de Pediatría; Argentina. Fil: Basquiera, Ana. Instituto Universitario de Ciencias Biomédicas de Córdoba; Argentina. Fil: Basquiera, Ana. Hospital Privado Universitario de Córdoba. Servicio de Oncohematologia; Argentina. Fil: Ricci, Brenda. Hospital Privado Universitario de Córdoba. Servicio de Oncohematologia; Argentina. Fil: Schmitz Abe, Klaus. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Schmitz Abe, Klaus. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Schmitz Abe, Klaus. Boston Children’s Hospital. The Manton Center for Orphan Disease Research, Boston; USA. Fil: Schmitz Abe, Klaus. University of Miami Miller School of Medicine and Holtz Children's Hospital Division of Neonatology. Department of Pediatrics. Jackson Health System, Miami; USA. Fil: Charbonnier, Louis-Marie. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Charbonnier, Louis-Marie. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Benamar, Mehdi. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Benamar, Mehdi. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Motrich, Rubén Darío. Universidad Nacional de Córdoba. Facultad de Ciencias Químicas. Departamento de Bioquímica Clínica; Argentina. Fil: Motrich, Rubén Darío. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro de Investigaciones en Bioquímica Clínica e Inmunología; Argentina. Fil: Motrich, Rubén Darío. Federation of Clinical Immunology Societies. Centro de Inmunología Clínica de Córdoba; Argentina. Fil: Chatila, Talal A. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Chatila, Talal A. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Rivero, Virginia E. Universidad Nacional de Córdoba. Facultad de Ciencias Químicas. Departamento de Bioquímica Clínica; Argentina. Fil: Rivero, Virginia E. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro de Investigaciones en Bioquímica Clínica e Inmunología; Argentina. Fil: Rivero, Virginia E. Federation of Clinical Immunology Societies. Centro de Inmunología Clínica de Córdoba; Argentina. Background: Common Variable Immunodeficiency (CVID) is the most common symptomatic syndrome among inborn errors of immunity (IEI). Although several aspects of CVID immunopathology have been elucidated, predictive factors for mortality are incompletely defined. A genetic cause can only be identified in approximately 30% of patients. Objectives: To develop a mortality predictive score based on the immunophenotypes and genotypes of CVID patients. Methods: Twenty-one patients diagnosed with CVID in Cordoba, Argentina, were analyzed for clinical and laboratory data. Immunophenotyping was done by flow cytometry. CVID-associated mutations were identified by whole exome sequencing (WES). Results: Live (15) and deceased (6) patients were compared. Univariate analysis showed significant differences in CD4+ T cells (p=0.002), Natural Killer (NK) cells (p=0.001) and memory switched B cells (p=0.001) between groups. Logistic regression analysis showed a negative correlation between CD4, NK and memory switched B cells counts and probability of survival over a 10-year period [CD4+ T cells: OR 1.01 (95% CI: 1.001-1,020); NK cells: OR 1.07 (95% CI:1.02-1.17) and memory switched B cells: OR 26.23 (95% CI: 2.06-2651.96). ROC curve analysis identified a survival cut off point for each parameter: CD4+ T cells 546 cell/ml AUC 0.87 (sensitivity 60%-specificity 100%), memory switched B cells 0.84 cells/ml AUC 0.92 (sensitivity 100%-specificity 85%), NK cells 45 cells/ml for AUC 0.92 (sensitivity 83%-specificity 100%), %). Genetic analysis on 14 patients from the cohort (9 females, 5 males) revealed mutations associated with IEI in 6 patients. Conclusions: A score to predict mortality is proposed based on CD4, NK and memory switched B cell number in patients with CVID. info:eu-repo/semantics/acceptedVersion Fil: Kahn, Adrián. Hospital Privado Universitario de Córdoba. Servicio de Alergia e Inmunología Clínica; Argentina. Fil: Kahn, Adrián. Instituto Universitario de Ciencias Biomédicas de Córdoba; Argentina. Fil: Kahn, Adrián. Federation of Clinical Immunology Societies. Centro de Inmunología Clínica de Córdoba; Argentina. Fil: Luque, Gabriela. Hospital Privado Universitario de Córdoba. Servicio de Oncohematologia; Argentina. Fil: Cuestas, Eduardo. Instituto Universitario de Ciencias Biomédicas de Córdoba; Argentina. Fil: Cuestas, Eduardo. Hospital Privado Universitario de Córdoba. Servicio de Pediatría; Argentina. Fil: Basquiera, Ana. Instituto Universitario de Ciencias Biomédicas de Córdoba; Argentina. Fil: Basquiera, Ana. Hospital Privado Universitario de Córdoba. Servicio de Oncohematologia; Argentina. Fil: Ricci, Brenda. Hospital Privado Universitario de Córdoba. Servicio de Oncohematologia; Argentina. Fil: Schmitz Abe, Klaus. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Schmitz Abe, Klaus. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Schmitz Abe, Klaus. Boston Children’s Hospital. The Manton Center for Orphan Disease Research, Boston; USA. Fil: Schmitz Abe, Klaus. University of Miami Miller School of Medicine and Holtz Children's Hospital Division of Neonatology. Department of Pediatrics. Jackson Health System, Miami; USA. Fil: Charbonnier, Louis-Marie. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Charbonnier, Louis-Marie. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Benamar, Mehdi. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Benamar, Mehdi. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Motrich, Rubén Darío. Universidad Nacional de Córdoba. Facultad de Ciencias Químicas. Departamento de Bioquímica Clínica; Argentina. Fil: Motrich, Rubén Darío. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro de Investigaciones en Bioquímica Clínica e Inmunología; Argentina. Fil: Motrich, Rubén Darío. Federation of Clinical Immunology Societies. Centro de Inmunología Clínica de Córdoba; Argentina. Fil: Chatila, Talal A. Boston Children’s Hospital. Division of Immunology, Boston; USA. Fil: Chatila, Talal A. Harvard Medical School. Department of Pediatrics, Boston; USA. Fil: Rivero, Virginia E. Universidad Nacional de Córdoba. Facultad de Ciencias Químicas. Departamento de Bioquímica Clínica; Argentina. Fil: Rivero, Virginia E. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro de Investigaciones en Bioquímica Clínica e Inmunología; Argentina. Fil: Rivero, Virginia E. Federation of Clinical Immunology Societies. Centro de Inmunología Clínica de Córdoba; Argentina. 2024-08-04T21:23:49Z 2024-08-04T21:23:49Z 2024-07-23 article Kahn A, Luque G, Cuestas E, Basquiera A, Ricci B, Schmitz-Abe K, Charbonnier L-M, Benamar M, Motrich RD, Chatila TA, Rivero VE, Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency, Journal of Allergy and Clinical Immunology: Global (2024), doi: https://doi.org/10.1016/ j.jacig.2024.100311. http://hdl.handle.net/11086/553072 1097-6825 https://www.sciencedirect.com/science/article/pii/S2772829324001073 https://www.jaci-global.org/article/S2772-8293(24)00107-3/fulltext https://doi.org/10.1016/j.jacig.2024.100311 eng Attribution-NonCommercial-NoDerivatives 4.0 International http://creativecommons.org/licenses/by-nc-nd/4.0/ American Academy of Allergy
spellingShingle CVID
Common variable immunodeficiency
Score
Mortality, Outcomes
WES
Whole exome sequencing
B cells subpopulations
T cells
Kahn, Adrián
Luque, Gabriela
Cuestas, Eduardo
Basquiera, Ana
Ricci, Brenda
Schmitz-Abe, Klaus
Charbonnier, Louis-Marie
Benamar, Mehdi
Motrich, Rubén Darío
Chatila, Talal A.
Rivero, Virginia E.
Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency
title Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency
title_full Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency
title_fullStr Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency
title_full_unstemmed Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency
title_short Immunological biomarkers associated with survival in a cohort of Argentinian patients with Common Variable Immunodeficiency
title_sort immunological biomarkers associated with survival in a cohort of argentinian patients with common variable immunodeficiency
topic CVID
Common variable immunodeficiency
Score
Mortality, Outcomes
WES
Whole exome sequencing
B cells subpopulations
T cells
url http://hdl.handle.net/11086/553072
https://www.sciencedirect.com/science/article/pii/S2772829324001073
https://www.jaci-global.org/article/S2772-8293(24)00107-3/fulltext
https://doi.org/10.1016/j.jacig.2024.100311
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