Lymphoma in Pediatric-Onset Inflammatory Bowel Disease Treated with Infliximab Monotherapy: A Case Series

Alejandro Llanos-Chea; Jason M Shapiro; Rachel W Winter; Logan Jerger; Timothy Menz; Meghan Gibson; Alison M Friedmann; Diana Treaba; Konstantinos Papamichael; Adam S Cheifetz; Sonia Friedman; Matthew J Hamilton; Harland S Winter

doi:10.1007/s10620-021-06884-9

Lymphoma in Pediatric-Onset Inflammatory Bowel Disease Treated with Infliximab Monotherapy: A Case Series

Dig Dis Sci. 2022 Jan;67(1):252-258. doi: 10.1007/s10620-021-06884-9. Epub 2021 Feb 17.

Authors

Alejandro Llanos-Chea^{1

2}, Jason M Shapiro^{3

4}, Rachel W Winter^{5

6}, Logan Jerger^{1

2}, Timothy Menz^{3

4}, Meghan Gibson^{3

4}, Alison M Friedmann^{1

2}, Diana Treaba^{7

8}, Konstantinos Papamichael^{9

10}, Adam S Cheifetz^{9

10}, Sonia Friedman^{5

6}, Matthew J Hamilton^{5

6}, Harland S Winter^{11

12}

Affiliations

¹ Pediatric Gastroenterology and Nutrition, Massachusetts General Hospital, CRPZ 5-560, 175 Cambridge Street, Boston, MA, 02114, USA.
² Harvard Medical School, CRPZ 5-560, 175 Cambridge Street, Boston, MA, 02114, USA.
³ Division of Pediatric Gastroenterology, Nutrition, and Liver Diseases, Hasbro Children's Hospital and Rhode Island Hospital, Hasbro Lower Level, 593 Eddy St., Providence, RI, 02903, USA.
⁴ Warren Alpert Medical School of Brown University, Hasbro Lower Level, 593 Eddy St., Providence, RI, 02903, USA.
⁵ Division of Gastroenterology, Brigham and Women's Hospital, Crohn's and Colitis Center, 75 Francis Street, Boston, MA, 02115, USA.
⁶ Harvard Medical School, 75 Francis Street, Boston, MA, 02115, USA.
⁷ Department of Pathology, Rhode Island Hospital, The Miriam Hospital, Brown University, Box G-E5, Providence, RI, 02912, USA.
⁸ Warren Alpert Medical School of Brown University, Brown University, Box G-E5, Providence, RI, 02912, USA.
⁹ Division of Gastroenterology, Beth Israel Deaconess Medical Center, Beth Israel Deaconess Med Ctr., 330 Brookline Avenue, Rabb Rose 1, Boston, MA, 02215, USA.
¹⁰ Harvard Medical School, Beth Israel Deaconess Med Ctr., 330 Brookline Avenue, Rabb Rose 1, Boston, MA, 02215, USA.
¹¹ Pediatric Gastroenterology and Nutrition, Massachusetts General Hospital, CRPZ 5-560, 175 Cambridge Street, Boston, MA, 02114, USA. hwinter@mgh.harvard.edu.
¹² Harvard Medical School, CRPZ 5-560, 175 Cambridge Street, Boston, MA, 02114, USA. hwinter@mgh.harvard.edu.

PMID: 33595782
DOI: 10.1007/s10620-021-06884-9

Abstract

Background: Inflammatory bowel diseases (IBD) are often treated with anti-tumor necrosis factor alpha (anti-TNFα) medications. Concomitant treatment of IBD with anti-TNFα agents and immunomodulators appears to be associated with an increased risk for lymphoma.

Methods: Patients who developed lymphoma while on monotherapy with an anti-TNFα agent were identified at three centers. Institutional Review Board approval was obtained.

Results: Five adolescents and young adult patients with pediatric-onset IBD who were treated with infliximab (IFX) without exposure to thiopurines were subsequently diagnosed with lymphoma. Three of the five patients had bone involvement at presentation. Epstein-Barr virus was positive in 2 cases. Median time from diagnosis of IBD and exposure to IFX prior to diagnosis of lymphoma was 5 and 4.3 years, respectively.

Conclusions: This case series reports long-term follow-up for young patients with IBD who were treated with IFX monotherapy and developed lymphoma. Three of the five patients had bone involvement. In general, the risk of lymphoma following exposure to anti-TNFα medications alone remains low, but the incidence of primary bone lymphomas in IBD has not been reported. Studies examining longer exposure times may be needed to determine the true lymphoma risk in patients treated with IFX monotherapy.

Keywords: ANTI-TNFα; Adolescent; Inflammatory bowel diseases; Infliximab; Lymphoma; Young adult.

Publication types

Case Reports
Research Support, Non-U.S. Gov't

MeSH terms

Adolescent
Age of Onset
Antineoplastic Combined Chemotherapy Protocols / administration & dosage*
Bone and Bones / diagnostic imaging
Bone and Bones / pathology
Colitis, Ulcerative* / drug therapy
Colitis, Ulcerative* / epidemiology
Crohn Disease* / drug therapy
Crohn Disease* / epidemiology
Drug Substitution / methods*
Female
Herpesvirus 4, Human / isolation & purification
Humans
Infliximab* / administration & dosage
Infliximab* / adverse effects
Lymph Nodes / diagnostic imaging
Lymph Nodes / pathology
Lymphoma* / diagnosis
Lymphoma* / etiology
Lymphoma* / physiopathology
Lymphoma* / therapy
Male
Treatment Outcome
Tumor Necrosis Factor Inhibitors / administration & dosage
Tumor Necrosis Factor Inhibitors / adverse effects
Young Adult

Substances

Tumor Necrosis Factor Inhibitors
Infliximab