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Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT

Rituximab (RTX) is an anti-CD20 monoclonal antibody that targets B cells—from the immature pre-B-cell stage in the bone marrow to mature circulating B cells—while preserving stem cells and plasma cells. It is used to treat autoimmune diseases, hematological malignancies, or complications after hemat...

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Autores principales: Luterbacher, Fanny, Bernard, Fanette, Baleydier, Frédéric, Ranza, Emmanuelle, Jandus, Peter, Blanchard-Rohner, Geraldine
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8727997/
https://www.ncbi.nlm.nih.gov/pubmed/35003091
http://dx.doi.org/10.3389/fimmu.2021.773853
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author Luterbacher, Fanny
Bernard, Fanette
Baleydier, Frédéric
Ranza, Emmanuelle
Jandus, Peter
Blanchard-Rohner, Geraldine
author_facet Luterbacher, Fanny
Bernard, Fanette
Baleydier, Frédéric
Ranza, Emmanuelle
Jandus, Peter
Blanchard-Rohner, Geraldine
author_sort Luterbacher, Fanny
collection PubMed
description Rituximab (RTX) is an anti-CD20 monoclonal antibody that targets B cells—from the immature pre-B-cell stage in the bone marrow to mature circulating B cells—while preserving stem cells and plasma cells. It is used to treat autoimmune diseases, hematological malignancies, or complications after hematopoietic stem cell transplantation (HSCT). Its safety profile is acceptable; however, a subset of patients can develop persistent hypogammaglobulinemia and associated severe complications, especially in pediatric populations. We report the unrelated cases of two young men aged 17 and 22, presenting with persistent hypogammaglobulinemia more than 7 and 10 years after treatment with RTX, respectively, and administered after HSCT for hemolytic anemia and Epstein–Barr virus reactivation, respectively. Both patients’ immunological workups showed low levels of total immunoglobulin, vaccine antibodies, and class switched-memory B cells but an increase in naive B cells, which can also be observed in primary immunodeficiencies such as those making up common variable immunodeficiency. Whole exome sequencing for one of the patients failed to detect a pathogenic variant causing a Mendelian immunological disorder. Annual assessments involving interruption of immunoglobulin replacement therapy each summer failed to demonstrate the recovery of endogenous immunoglobulin production or normal numbers of class switched-memory B cells 7 and 10 years after the patients’ respective treatments with RTX. Although the factors that may lead to prolonged hypogammaglobulinemia after rituximab treatment (if necessary) remain unclear, a comprehensive immunological workup before treatment and long-term follow-up are mandatory to assess long-term complications, especially in children.
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spelling pubmed-87279972022-01-06 Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT Luterbacher, Fanny Bernard, Fanette Baleydier, Frédéric Ranza, Emmanuelle Jandus, Peter Blanchard-Rohner, Geraldine Front Immunol Immunology Rituximab (RTX) is an anti-CD20 monoclonal antibody that targets B cells—from the immature pre-B-cell stage in the bone marrow to mature circulating B cells—while preserving stem cells and plasma cells. It is used to treat autoimmune diseases, hematological malignancies, or complications after hematopoietic stem cell transplantation (HSCT). Its safety profile is acceptable; however, a subset of patients can develop persistent hypogammaglobulinemia and associated severe complications, especially in pediatric populations. We report the unrelated cases of two young men aged 17 and 22, presenting with persistent hypogammaglobulinemia more than 7 and 10 years after treatment with RTX, respectively, and administered after HSCT for hemolytic anemia and Epstein–Barr virus reactivation, respectively. Both patients’ immunological workups showed low levels of total immunoglobulin, vaccine antibodies, and class switched-memory B cells but an increase in naive B cells, which can also be observed in primary immunodeficiencies such as those making up common variable immunodeficiency. Whole exome sequencing for one of the patients failed to detect a pathogenic variant causing a Mendelian immunological disorder. Annual assessments involving interruption of immunoglobulin replacement therapy each summer failed to demonstrate the recovery of endogenous immunoglobulin production or normal numbers of class switched-memory B cells 7 and 10 years after the patients’ respective treatments with RTX. Although the factors that may lead to prolonged hypogammaglobulinemia after rituximab treatment (if necessary) remain unclear, a comprehensive immunological workup before treatment and long-term follow-up are mandatory to assess long-term complications, especially in children. Frontiers Media S.A. 2021-12-22 /pmc/articles/PMC8727997/ /pubmed/35003091 http://dx.doi.org/10.3389/fimmu.2021.773853 Text en Copyright © 2021 Luterbacher, Bernard, Baleydier, Ranza, Jandus and Blanchard-Rohner https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Immunology
Luterbacher, Fanny
Bernard, Fanette
Baleydier, Frédéric
Ranza, Emmanuelle
Jandus, Peter
Blanchard-Rohner, Geraldine
Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT
title Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT
title_full Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT
title_fullStr Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT
title_full_unstemmed Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT
title_short Case Report: Persistent Hypogammaglobulinemia More Than 10 Years After Rituximab Given Post-HSCT
title_sort case report: persistent hypogammaglobulinemia more than 10 years after rituximab given post-hsct
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8727997/
https://www.ncbi.nlm.nih.gov/pubmed/35003091
http://dx.doi.org/10.3389/fimmu.2021.773853
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