Clinical Case Summary

Introduction: Tofacitinib is an oral small molecule JAK inhibitor approved for the treatment of moderate to severe ulcerative colitis (UC). Its efficacy and safety have been demonstrated in phase III clinical trials and supported by real-life data.
Case description: We report the case of an 18-year-old woman with a 1-year diagnosis of left-sided UC, with multiple admissions due to disease exacerbation or infections, refractory to infliximab (with azathioprine) and currently under treatment with vedolizumab and tacrolimus. She was admitted due to a severe disease exacerbation and, because of a previous history of neuropsychiatric side effects to corticotherapy, tofacitinib was initiated. In the following 6 days, there was no clinical improvement of UC, and serial blood work-up revealed moderate grade persistent peripheral eosinophilia (3000 cells/mm3) and acute kidney injury grade 1 KDIGO. Tofacitinib temporary suspension was decided, with a rapid normalization of renal function/eosinophil levels. Tofacitinib was restarted 2 days after its suspension. However, she developed moderate eosinophilia (2000 cells/mm3) again, which was considered an adverse effect (AE) to tofacitinib, leading to its suspension with eosinophilia resolution. Given the severity of the disease, after a multidisciplinary discussion, it was decided to start high-dose corticotherapy and ustekinumab with maintenance therapy every 4 weeks, and to add tacrolimus. Clinical and biochemical remission were achieved, and the patient was discharged. Three-month follow-up after tofacitinib suspension showed no recrudescence of eosinophilia.
Conclusion: Tofacitinib represents a significant advance in the management of UC patients. The drug has a good safety profile with few related AE. This case aims to warn about type IVb hypersensitivity reaction as an AE of tofacitinib not reported so far. To our knowledge this is the first case of tofacitinib-induced eosinophilia in the context of UC.

Introduction

​​​​​Concerns regarding colorectal cancer in short bowel syndrome (SBS) patients receiving teduglutide (TED) treatment have led to the recommendation of colonoscopy for screening and surveillance.^1,2 While there are reports of new small bowel polyps in TED patients, data on small bowel cancer risk and surveillance are lacking.^3,4 This study aims to evaluate the feasibility and effectiveness of using double-balloon enteroscopy (DBE) for surveillance in SBS patients undergoing TED treatment.

Aims & Methods

Data from patients with SBS in TED treatment who underwent DBE within a year of starting TED at a tertiary referral center for enteroscopy between December 2019 and October 2023 were collected. The primary endpoints were technical success, adverse events, and total enteroscopy rates. Secondary endpoints included the incidence of small bowel polyps and residual small bowel length measurement.

Results

Nine patients (7 female, median age 65.5, IQR 58-79) with SBS (22% type 1, 55.6% type 2, 22.2% type 3-SBS) underwent DBE (10 anterograde, 2 retrograde) under procedural sedation. Crohn’s disease (33.3%) and post-surgical complications (33.3%) were the most frequent causes of SBS. The median interval from TED prescription to DBE surveillance was 9.1 months (IQR 3.2-32.9). The median depth of maximal insertion was 85 cm (IQR 50-187). No adverse events occurred during all the procedures. The total enteroscopy rate was 75% (80% [8/10] of anterograde DBEs, 50% [1/2] of retrograde DBEs). Panenteric DBE allowed the estimation of small bowel residual length in all cases (2/2) where this important clinical information was unknown and revealed a different residual small bowel length compared to surgery in 44% of patients. No small bowel malignancies were detected during the study period.

Conclusion

DBE surveillance for small bowel malignancy in SBS patients undergoing TED treatment is feasible and effective. Total enteroscopy can be frequently achieved with a single endoscopy in these patients, allowing easy oncologic surveillance.

References

1. Revestive (teduglutide). Summary of product characteristics. Takeda; August 2022. Accessed 23 March 2024. https://www. medicines.org.uk/emc/product/3382/smpc
2. Pironi L, Allard JP, Joly F, et al. Use of teduglutide in adults with short bowel syndrome-associated intestinal failure. Nutr Clin Pract 2024;39:141-153.
3. Pevny S, Pape UF, Elezkurtaj S, et al. De Novo Development of Distal Jejunal and Duodenal Adenomas After 41 Months of Teduglutide Treatment in a Patient With Short-Bowel Syndrome: A Case Report. JPEN J Parenter Enteral Nutr 2021;45:652-656.
4. Ukleja A, Alkhairi B, Bejarano P, et al. De Novo Development of Hamartomatous Duodenal Polyps in a Patient With Short Bowel Syndrome During Teduglutide Therapy: A Case Report. JPEN J Parenter Enteral Nutr 2018;42:658-660.