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Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies

Children and adolescents with inflammatory bowel disease are often treated with immunomodulators (thiopurines, methotrexate) and biologics (anti-TNF, anti-integrin) for extended periods despite concerns about long-term safety. Here, we report a case of follicular dendritic cell sarcoma, a very rare...

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Autores principales: Anderson, Kaitlyn, Moss, Kerry, Campbell, Brendan, Moote, Douglas, Kakazu, Kari, Hyams, Jeffrey S.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins, Inc. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10158454/
https://www.ncbi.nlm.nih.gov/pubmed/37168632
http://dx.doi.org/10.1097/PG9.0000000000000231
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author Anderson, Kaitlyn
Moss, Kerry
Campbell, Brendan
Moote, Douglas
Kakazu, Kari
Hyams, Jeffrey S.
author_facet Anderson, Kaitlyn
Moss, Kerry
Campbell, Brendan
Moote, Douglas
Kakazu, Kari
Hyams, Jeffrey S.
author_sort Anderson, Kaitlyn
collection PubMed
description Children and adolescents with inflammatory bowel disease are often treated with immunomodulators (thiopurines, methotrexate) and biologics (anti-TNF, anti-integrin) for extended periods despite concerns about long-term safety. Here, we report a case of follicular dendritic cell sarcoma, a very rare malignancy, and the first reported presentation in a patient with inflammatory bowel disease exposed to infliximab, methotrexate, and vedolizumab. We review the key clinical features and diagnostic factors of this malignancy. The pathogenesis of follicular dendritic cell sarcoma is largely unknown, however, knock out of B-cell TNF in mice has been related to follicular dendritic cell dysregulation through its impact on NF-κB pathways and CXCL13 chemokines. It is unknown whether any relationship exists between this patient’s diagnosis of Crohn’s disease and therapeutic exposures to this rare malignancy. We document this case in the literature to raise awareness among other clinicians who may observe a similar case.
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spelling pubmed-101584542023-05-09 Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies Anderson, Kaitlyn Moss, Kerry Campbell, Brendan Moote, Douglas Kakazu, Kari Hyams, Jeffrey S. JPGN Rep Case Report Children and adolescents with inflammatory bowel disease are often treated with immunomodulators (thiopurines, methotrexate) and biologics (anti-TNF, anti-integrin) for extended periods despite concerns about long-term safety. Here, we report a case of follicular dendritic cell sarcoma, a very rare malignancy, and the first reported presentation in a patient with inflammatory bowel disease exposed to infliximab, methotrexate, and vedolizumab. We review the key clinical features and diagnostic factors of this malignancy. The pathogenesis of follicular dendritic cell sarcoma is largely unknown, however, knock out of B-cell TNF in mice has been related to follicular dendritic cell dysregulation through its impact on NF-κB pathways and CXCL13 chemokines. It is unknown whether any relationship exists between this patient’s diagnosis of Crohn’s disease and therapeutic exposures to this rare malignancy. We document this case in the literature to raise awareness among other clinicians who may observe a similar case. Lippincott Williams & Wilkins, Inc. 2022-07-18 /pmc/articles/PMC10158454/ /pubmed/37168632 http://dx.doi.org/10.1097/PG9.0000000000000231 Text en Copyright © 2022 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and the North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY) (https://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Anderson, Kaitlyn
Moss, Kerry
Campbell, Brendan
Moote, Douglas
Kakazu, Kari
Hyams, Jeffrey S.
Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies
title Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies
title_full Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies
title_fullStr Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies
title_full_unstemmed Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies
title_short Follicular Dendritic Cell Sarcoma in a Patient With Adolescent-Onset Crohn’s Disease Exposed to Multiple Immunomodulator and Biologic Therapies
title_sort follicular dendritic cell sarcoma in a patient with adolescent-onset crohn’s disease exposed to multiple immunomodulator and biologic therapies
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10158454/
https://www.ncbi.nlm.nih.gov/pubmed/37168632
http://dx.doi.org/10.1097/PG9.0000000000000231
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