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Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease?

OBJECTIVE: To investigate the immunologic impact of a single cycle of rituximab (RTX) in children and adolescents with immune-mediated disorders, we evaluated B cells and immunoglobulin levels of 20 patients with neuroimmunologic, nephrologic, dermatologic, and rheumatologic disorders treated under...

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Autores principales: Deyà-Martínez, Angela, Gordón, Yadira, Molina-Anguita, Cristina, Vlagea, Alexandru, Piquer, Monica, Juan, Manel, Esteve-Solé, Ana, Antón, Jordi, Madrid, Álvaro, García-García, Ana, Plaza, Ana M., Armangue, Thaís, Alsina, Laia
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7217658/
https://www.ncbi.nlm.nih.gov/pubmed/32376706
http://dx.doi.org/10.1212/NXI.0000000000000724
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author Deyà-Martínez, Angela
Gordón, Yadira
Molina-Anguita, Cristina
Vlagea, Alexandru
Piquer, Monica
Juan, Manel
Esteve-Solé, Ana
Antón, Jordi
Madrid, Álvaro
García-García, Ana
Plaza, Ana M.
Armangue, Thaís
Alsina, Laia
author_facet Deyà-Martínez, Angela
Gordón, Yadira
Molina-Anguita, Cristina
Vlagea, Alexandru
Piquer, Monica
Juan, Manel
Esteve-Solé, Ana
Antón, Jordi
Madrid, Álvaro
García-García, Ana
Plaza, Ana M.
Armangue, Thaís
Alsina, Laia
author_sort Deyà-Martínez, Angela
collection PubMed
description OBJECTIVE: To investigate the immunologic impact of a single cycle of rituximab (RTX) in children and adolescents with immune-mediated disorders, we evaluated B cells and immunoglobulin levels of 20 patients with neuroimmunologic, nephrologic, dermatologic, and rheumatologic disorders treated under recommended guidelines. METHODS: Retrospective study of immunologic changes in children (aged ≤18 years) diagnosed with immune-mediated disorders in which RTX was prescribed between June 2014 and February 2019. Patients were excluded if they had prior diagnosis of malignant disease or primary immunodeficiency. Patients were clinically and immunologically followed up every 3 months. Only patients having received a single cycle of RTX and with a follow-up greater than 12 months were included in the analysis of persistent dysgammaglobulinemia. RESULTS: Twenty children were included. Median age at RTX treatment was 12.8 years (interquartile range [IQR] 6.6–15.5 years). Median follow-up was 12.6 months (IQR 10.2–24 months). Of the 14 patients eligible for persistent dysgammaglobulinemia analysis (3 had received RTX retreatment, 2 had <12 months post-RTX follow-up, and in 1 data for this time point was missing), 2/14 (14%) remained with complete B-cell depletion, and 5/14 (36%) had dysgammaglobulinemia. Patients with dysgammaglobulinemia were younger (7.8 vs 15.6 years, p = 0.072), had more underlying neuroimmunologic diseases (5/5 vs 0/9, p < 0.001), and had received more frequently concentrated doses of RTX (3/5 vs 1/9, p = 0.05) than patients without dysgammaglobulinemia. Kinetics of immunoglobulins in the 20 patients revealed a decrease as early as 3 months after RTX in patients with neuroimmunologic disorders. CONCLUSION: In our cohort, single-cycle RTX-induced dysgammaglobulinemia was enhanced in patients with neuroimmunologic diseases. Further studies are needed to confirm this observation.
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spelling pubmed-72176582020-06-02 Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease? Deyà-Martínez, Angela Gordón, Yadira Molina-Anguita, Cristina Vlagea, Alexandru Piquer, Monica Juan, Manel Esteve-Solé, Ana Antón, Jordi Madrid, Álvaro García-García, Ana Plaza, Ana M. Armangue, Thaís Alsina, Laia Neurol Neuroimmunol Neuroinflamm Article OBJECTIVE: To investigate the immunologic impact of a single cycle of rituximab (RTX) in children and adolescents with immune-mediated disorders, we evaluated B cells and immunoglobulin levels of 20 patients with neuroimmunologic, nephrologic, dermatologic, and rheumatologic disorders treated under recommended guidelines. METHODS: Retrospective study of immunologic changes in children (aged ≤18 years) diagnosed with immune-mediated disorders in which RTX was prescribed between June 2014 and February 2019. Patients were excluded if they had prior diagnosis of malignant disease or primary immunodeficiency. Patients were clinically and immunologically followed up every 3 months. Only patients having received a single cycle of RTX and with a follow-up greater than 12 months were included in the analysis of persistent dysgammaglobulinemia. RESULTS: Twenty children were included. Median age at RTX treatment was 12.8 years (interquartile range [IQR] 6.6–15.5 years). Median follow-up was 12.6 months (IQR 10.2–24 months). Of the 14 patients eligible for persistent dysgammaglobulinemia analysis (3 had received RTX retreatment, 2 had <12 months post-RTX follow-up, and in 1 data for this time point was missing), 2/14 (14%) remained with complete B-cell depletion, and 5/14 (36%) had dysgammaglobulinemia. Patients with dysgammaglobulinemia were younger (7.8 vs 15.6 years, p = 0.072), had more underlying neuroimmunologic diseases (5/5 vs 0/9, p < 0.001), and had received more frequently concentrated doses of RTX (3/5 vs 1/9, p = 0.05) than patients without dysgammaglobulinemia. Kinetics of immunoglobulins in the 20 patients revealed a decrease as early as 3 months after RTX in patients with neuroimmunologic disorders. CONCLUSION: In our cohort, single-cycle RTX-induced dysgammaglobulinemia was enhanced in patients with neuroimmunologic diseases. Further studies are needed to confirm this observation. Lippincott Williams & Wilkins 2020-05-06 /pmc/articles/PMC7217658/ /pubmed/32376706 http://dx.doi.org/10.1212/NXI.0000000000000724 Text en Copyright © 2020 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology. This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License 4.0 (CC BY-NC-ND) (http://creativecommons.org/licenses/by-nc-nd/4.0/) , which permits downloading and sharing the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.
spellingShingle Article
Deyà-Martínez, Angela
Gordón, Yadira
Molina-Anguita, Cristina
Vlagea, Alexandru
Piquer, Monica
Juan, Manel
Esteve-Solé, Ana
Antón, Jordi
Madrid, Álvaro
García-García, Ana
Plaza, Ana M.
Armangue, Thaís
Alsina, Laia
Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease?
title Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease?
title_full Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease?
title_fullStr Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease?
title_full_unstemmed Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease?
title_short Single-cycle rituximab-induced immunologic changes in children: Enhanced in neuroimmunologic disease?
title_sort single-cycle rituximab-induced immunologic changes in children: enhanced in neuroimmunologic disease?
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7217658/
https://www.ncbi.nlm.nih.gov/pubmed/32376706
http://dx.doi.org/10.1212/NXI.0000000000000724
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