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

Abstract

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.

Algorithmic Identification of Treatment‐Emergent Adverse Events From Clinical Notes Using Large Language Models: A Pilot Study in Inflammatory Bowel Disease

“…For instance, our dataset included a patient receiving an anti-TNF who was hospitalized for a new diagnosis of sarcoma. Sarcomas have previously been reported in the context of children with IBD using anti-TNFs, 24 although multiple long-term observational studies have not consistently found a link between anti-TNF use and an increased risk of cancer in adults. 25,26 Non-steroid medications for IBD are not uniformly effective.…”

Algorithmic identification of treatment-emergent adverse events from clinical notes using large language models: a pilot study in inflammatory bowel disease

“…For instance, our dataset included a patient receiving an anti-TNF who was hospitalized for a new diagnosis of sarcoma. Sarcomas have previously been reported in the context of children with IBD using anti-TNFs 24 , although multiple long-term observational studies have not consistently found a link between anti-TNF use and an increased risk of cancer in adults 25,26 . Future directions of this work include external validation using data from additional centers, expansion to additional disease states outside of IBD, and downstream studies designed to identify new drug-SAEs associations more rigorously using aggregated data.…”