Available online 10 October 2022

Abstract

Crohn’s disease can be characterized as a chronic inflammatory state causing various clinical presentations and long-term risks that should be considered when determining the optimal therapeutic strategy. To date, while a few case reports have been available regarding ustekinumab-induced thrombocytopenia, none are available regarding hypersplenism. We describe a 33-year-old woman who developed only Ileocolonic Crohn’s disease on ustekinumab due to failure of anti-TNF with septic shock and thrombocytopenia. Abdominal computed tomography revealed hepatosplenomegaly, parasacral collection, and fistulization. The patient was transferred to the intensive care unit and managed accordingly. Various treatment modalities were attempted, but none of them improved her platelet count. Our case report demonstrates that ustekinumab may induce hypersplenism and subsequently thrombocytopenia and should be considered a potential cause of low platelet count.

Introduction

Crohn’s disease can be characterized as a chronic inflammatory condition affecting the gastrointestinal tract. Around 6%–46% of affected individuals experience extraintestinal manifestations [1], [2]. However, no published data have been available regarding hypersplenism in Crohn's disease.

Nowadays, different medications have been used to achieve and subsequently maintain remission, among which is ustekinumab, a monoclonal antibody targeting the p40 subunit of interleukin 12 and interleukin 23, which is a key cytokine in the pathogenesis of Crohn’s disease [3]. In 2016, ustekinumab was approved by the US FDA for treating Crohn’s disease [4]. In line with this, a study by Wiliam et al. investigated the safety profile of ustekinumab in an inflammatory bowel disease population over a 1-year duration and found that it was favorable and comparable to placebo [5].

Well-known side effects are associated with ustekinumab. However, to the best of our knowledge, no case reports have been published regarding ustekinumab-induced hypersplenism. In this case report, we sought to determine the association between ustekinumab and the development of hypersplenism in patients with Crohn’s disease.

Section snippetsCase presentation

We report the case of a 33-year-old woman who had only Ileocolonic Crohn’s disease diagnosed in 2004 with a history of multiple resections and ileorectal anastomosis with an ileocolonic fistula.

The patient presented to the emergency department with a 10-day history of bilateral flank pain and fever. Before her current presentation, the patient received azathioprine and infliximab. Infliximab was initiated in 2015 and discontinued in 2020, with no development of antibodies.

Her latest colonoscopy

Discussion

In this report, we present the case of a woman, with a history of only Crohn’s disease receiving ustekinumab, who developed hypersplenism and subsequent severe thrombocytopenia. This presentation agrees with secondary hypersplenism and is the first report to show an association between hypersplenism and ustekinumab.

The introduction of monoclonal antibodies, such as ustekinumab, has changed the therapeutic strategy of Crohn’s disease in the last decade. Whereby it targets the p40 subunit of

Declaration of competing interests

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

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© 2022 Pan-Arab Association of Gastroenterology. Published by Elsevier B.V. All rights reserved.