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Copyright ©The Author(s) 2025. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 16, 2025; 13(35): 114672
Published online Dec 16, 2025. doi: 10.12998/wjcc.v13.i35.114672
Endometrial stromal sarcoma in a patient with ulcerative colitis receiving immunosuppressive therapy: A case report and review of literature
Ada Alexandrina Brom dos Santos Soares, Thais Gagno Grillo, Lívia Cafundó Almeida, Gabriel S Thiago Cavalleiro, Marina Amorim Lopes, Marcela Maria Silvino Craveiro, Giedre Soares Prates Herrerias, Júlio Pinheiro Baima, Rogerio Saad-Hossne, Ligia Yukie Sassaki
Ada Alexandrina Brom dos Santos Soares, Thais Gagno Grillo, Lívia Cafundó Almeida, Gabriel S Thiago Cavalleiro, Giedre Soares Prates Herrerias, Júlio Pinheiro Baima, Ligia Yukie Sassaki, Department of Internal Medicine, Medical School, São Paulo State University (UNESP), Botucatu 18618687, São Paulo, Brazil
Marina Amorim Lopes, Marcela Maria Silvino Craveiro, Rogerio Saad-Hossne, Department of Surgery, Medical School, São Paulo State University (UNESP), Botucatu 18618687, São Paulo, Brazil
Author contributions: All authors contributed to this manuscript. Soares AABS, Grillo TG, Almeida LC, Cavalleiro GST, Lopes MA, Craveiro MMS, Herrerias GSP, Baima JP, Saad-Hossne R, Sassaki LY contributed to the conception and design of the study; the acquisition, analysis, and interpretation of data; drafting the article, revising it critically for important intellectual content, and approving the final version to be submitted.
Informed consent statement: Written informed consent was obtained from the patient for publication of this case report and any accompanying images.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Thais Gagno Grillo, PhD, Department of Internal Medicine, Medical School, São Paulo State University (UNESP), Av. Prof. Mário Rubens Guimarães Montenegro, Botucatu 18618687, São Paulo, Brazil. thais.gagno@unesp.br
Received: September 28, 2025
Revised: October 25, 2025
Accepted: December 1, 2025
Published online: December 16, 2025
Processing time: 80 Days and 7.4 Hours
Abstract
BACKGROUND

Ulcerative colitis (UC) is a chronic inflammatory bowel disease treated with immunosuppressants to control inflammation. Drugs like azathioprine (AZA) and anti-tumor necrosis factor agents increase the risk of extraintestinal malignancies. However, no association has been established between these therapies and endometrial stromal sarcoma. This report presents a rare case of endometrial stromal sarcoma in a patient with UC undergoing immunosuppressive treatment and includes a literature review to explore any possible correlation between the disease, the therapies used, and the development of this rare tumor.

CASE SUMMARY

Female, 49 years old, with UC pancolitis extension since 2017. She used aminosalicylates and AZA with non-response. She started infliximab and AZA combination therapy in 2020, with optimization in 2021 due to endoscopic activity. In the same year, the patient presented to the emergency room with ascitis and underwent diagnostic paracentesis, which showed serum-ascites albumin gradient < 1.1 g/dL, absence of neoplastic cells, and abdominal and pelvic tomography reported a hypoechoic nodular lesion in the posterior wall of the uterus and elevated carbohydrate antigen 125. Given the suspicion of neoplasia, the suspension of immunosuppressive therapy was indicated. The patient underwent a total abdominal hysterectomy and bilateral salpingo-oophorectomy, and the pathological result reported low-grade endometrial sarcoma. It was decided to introduce vedolizumab for the management of UC; however, even after induction therapy, intense clinical and endoscopic disease activity was maintained, with total proctocolectomy being indicated due to clinical refractoriness and a history of neoplasia.

CONCLUSION

Patients with inflammatory bowel disease have a higher risk of cancer due to inflammation or treatment. Proper screening with multidisciplinary care can improve outcomes.

Keywords: Ulcerative colitis; Endometrial stromal sarcoma; Azathioprine; Infliximab; Inflammatory bowel disease; Case report

Core Tip: Ulcerative colitis is a chronic inflammatory bowel disease treated with immunosuppressants. Though rare, treatments such as azathioprine and anti-tumor necrosis factor therapy may increase the risk of extraintestinal cancers. This report describes a patient with ulcerative colitis receiving immunosuppressive therapy who was diagnosed with endometrial stromal sarcoma during treatment. Clinical, diagnostic, imaging, and treatment details are discussed. The case highlights the importance of understanding medication side effects to enhance patient safety, prognosis, and quality of life.