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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...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Lippincott Williams & Wilkins, Inc.
2022
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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. |
format | Online Article Text |
id | pubmed-10158454 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Lippincott Williams & Wilkins, Inc. |
record_format | MEDLINE/PubMed |
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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