Conservative Management Achieving Durable Clinical and Radiologic Remission in Adolescent PSC-UC Overlap Without Biologic Therapy

Conservative Management Achieving Durable Clinical and Radiologic Remission in Adolescent PSC-UC Overlap Without Biologic Therapy

Ryan Evan Bruner, B.A., B.S *1, Osama AlOudat, M.D 1, Monica Rao, B.S 1, Aniruddh Setya, M.D 1

 

  1. Saint Louis University, SSM Health Cardinal Glennon Children’s Hospital, Department of Pediatric Gastroenterology, St. Louis, MO.


*Correspondence to: Ryan Evan Bruner, Saint Louis University, SSM Health Cardinal Glennon Children’s Hospital, Department of Pediatric Gastroenterology, St. Louis, MO.


Copyright

© 2026 Ryan Evan Bruner, This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Received: 24 March 2026

Published: 01 April 2026

DOI: https://doi.org/10.5281/zenodo.19367314

 

Abstract

Pediatric primary sclerosing cholangitis (PSC) associated with ulcerative colitis (UC) presents unique diagnostic and therapeutic challenges. Conventional management frequently involves biologic agents due to disease severity.

We present a 17-year-old male diagnosed with PSC-UC overlap syndrome who achieved sustained clinical and radiologic remission using conservative therapy comprising mesalamine, corticosteroids, and microbiome modulation with oral vancomycin. Despite initial concerns of disease progression indicated by elevated liver enzymes, adherence to conservative therapy led to substantial clinical improvement without escalation to biologics. Magnetic resonance cholangiopancreatography (MRCP) confirmed radiologic remission, and subsequent follow-up revealed consistent clinical stability and weight gain after addressing medication adherence and supplement misuse.

This case underscores the possibility of achieving durable remission in adolescent PSC-UC overlap through early diagnosis, adherence to conservative therapy, and close multidisciplinary monitoring, thus potentially avoiding biologic-associated risks.


Conservative Management Achieving Durable Clinical and Radiologic Remission in Adolescent PSC-UC Overlap Without Biologic Therapy

Introduction

Primary sclerosing cholangitis (PSC) is a chronic cholestatic liver disease characterized by progressive inflammation and fibrosis of the bile ducts, frequently associated with ulcerative colitis (UC). The incidence of primary sclerosing cholangitis-ulcerative colitis (PSC-UC) overlap in pediatric patients is best described by the proportion of children with PSC who also have IBD, particularly UC. According to the American Association for the Study of Liver Diseases, IBD is present in approximately 63–80% of pediatric PSC cases, and more than two-thirds of these cases are ulcerative colitis, making the overlap of PSC-UC the most common phenotype in this population.[1] Pediatric PSC remains rare and presents distinct management challenges due to its variable progression and limited therapeutic guidelines. Although biologic therapies are increasingly used, they pose significant risks, including immunosuppression and high costs. Conservative management, including mesalamine, corticosteroids, and microbiota-directed therapies such as oral vancomycin, presents an attractive alternative. This report highlights successful conservative management of PSC-UC overlap in adolescence, emphasizing therapeutic potential and clinical outcomes without biologics.

 

Case Presentation

A 17-year-old Caucasian male presented with symptoms suggestive of an ulcerative colitis flare, including significant weight loss, mild abdominal discomfort, and two episodes of non-bloody diarrhea per day. Initially diagnosed with ulcerative pancolitis in December 2022, he subsequently received a PSC diagnosis in January 2023 after MRCP demonstrated typical bile duct changes and significantly elevated gamma-glutamyl transferase (GGT) levels at 701 U/L (Figure 1). Treatment consisted of high-dose mesalamine (4.8g daily), oral vancomycin (250mg four times daily), and corticosteroids (prednisone taper starting at 40mg).

Clinical improvement was initially achieved, as evidenced by normalized GGT levels (76 U/L) and radiologic remission on follow-up MRCP. However, adherence issues arose from unsupervised intermittent prednisone use. Additionally, the patient consumed bodybuilding supplements (approximately 5 grams of creatine monohydrate) daily, leading to renal concerns reflected by elevated creatinine levels (1.18 mg/dL) during subsequent evaluation. Upon admission in January 2025, extensive counseling sessions addressing proper steroid use, supplement moderation, and medication adherence significantly improved patient compliance and resolved renal concerns, with creatinine levels decreasing to 1.09 mg/dL. The patient maintained stable remission, thereafter, confirmed clinically and by follow-up colonoscopy.

Axial (left) and coronal (right) T2-weighted MR images demonstrate mild beading of the central intrahepatic and extrahepatic bile ducts (arrows), with the common bile duct measuring up to 0.5 cm in diameter. Subtle wall thickening of the common bile duct is suspected. The gallbladder appears normal.

This table outlines the patient’s diagnostic timeline, laboratory changes, treatment interventions, and clinical outcomes throughout the course of PSC–UC overlap management. It highlights biochemical improvement following conservative therapy, the impact of adherence counseling, and sustained remission without the need for biologic therapy.

 

Discussion

This case illustrates a successful conservative therapeutic approach in managing pediatric PSC-UC overlap, a condition that typically poses significant challenges due to the complex interplay between hepatic and gastrointestinal inflammatory processes. Prior literature has demonstrated that the microbiota in PSC-UC is distinct from that of non-disease controls, PSC, or IBD phenotypes, and these changes may trigger changes in bile acid homeostasis and the inflammatory cycle that eventually causes bile duct fibrosis and strictures [2, 3]. Oral vancomycin played a central role in this conservative strategy by modulating the gut microbiome and reducing pathogenic bacterial populations, which may help reduce proinflammatory secondary bile acids, and exerting immunomodulatory effects by decreased production of the inflammatory cytokine TNF- α and increased production of the regulatory TGF- β 4-6. This microbiota-targeted approach aligns well with current understanding of the pathophysiology of PSC-UC, where dysbiosis and immune dysregulation are key components driving disease progression [2, 3, 7]. 

The patient's adherence to therapy emerged as a crucial factor in achieving and maintaining remission. Notably, this case highlights significant adherence challenges, including unsupervised steroid usage and misuse of bodybuilding supplements. While creatine monohydrate is recognized as safe in adult populations, there is a lack of placebo-controlled, blinded RCTs demonstrating its safety in the pediatric and pediatric chronic-disease populations [8]. Such scenarios underscore the importance of robust patient and caregiver education to prevent medication misuse and ensure proper adherence to prescribed therapies [5]. Education and multidisciplinary interventions in managing chronic pediatric illnesses have been shown to enhance therapeutic outcomes significantly, emphasizing the need for thorough discussions about medication regimens, side effects, and lifestyle modifications [6, 9, 10].

The avoidance of biologic therapy in this patient offers important clinical insights. While biologics represent potent therapeutic options, their associated risks—including increased susceptibility to infections, immune suppression, and substantial financial and psychological burdens—necessitate careful consideration, particularly in pediatric patients. Successfully managing pediatric PSC-UC with conservative therapy may mitigate these risks, improve quality of life, and promote better long-term outcomes [6].

 

Conclusion

This case reinforces the potential efficacy of individualized conservative management, combining microbiome-directed therapies, corticosteroids, and patient adherence education. It emphasizes the critical role of a personalized, multidisciplinary approach to pediatric PSC-UC management, potentially avoiding biologic therapies and improving patient outcomes. Informed consent was obtained from the patient and his father regarding publication of this case.

 

References

1. Chapman R, Fevery J, Kalloo A, et al. Diagnosis and management of primary sclerosing cholangitis. Hepatology 2010;51:660-78.

2. Liwinski T, Zenouzi R, John C, et al. Alterations of the bile microbiome in primary sclerosing cholangitis. Gut 2020;69:665-672.

3. Sannaa W, Almasry M, Peedikayil M, et al. Effectiveness and safety of oral vancomycin for the treatment of inflammatory bowel disease associated with primary sclerosing cholangitis: a systematic review and pooled analysis. Therapeutic Advances in Gastroenterology 2025;18:17562848241312766.

4. van Rheenen PF, Kolho KL, Russell RK, et al. Primary sclerosing cholangitis in children with inflammatory bowel disease: An ESPGHAN position paper from the Hepatology Committee and the IBD Porto group. J Pediatr Gastroenterol Nutr 2025;80:374-393.

5. Abarbanel DN, Seki SM, Davies Y, et al. Immunomodulatory effect of vancomycin on Treg in pediatric inflammatory bowel disease and primary sclerosing cholangitis. J Clin Immunol 2013;33:397-406.

6. Yoshimura A, Wakabayashi Y, Mori T. Cellular and molecular basis for the regulation of inflammation by TGF-beta. J Biochem 2010;147:781-92.

7. Shah A, Macdonald GA, Morrison M, et al. Targeting the Gut Microbiome as a Treatment for Primary Sclerosing Cholangitis: A Conceptional Framework. Official journal of the American College of Gastroenterology | ACG 2020;115.

8. Jagim AR, Kerksick CM. Creatine Supplementation in Children and Adolescents. Nutrients 2021;13.

9. McGrady ME, Hommel KA. Medication adherence and health care utilization in pediatric chronic illness: a systematic review. Pediatrics 2013;132:730-740.

10. Yao T-C, Wang J-Y, Chang S-M, et al. Association of Oral Corticosteroid Bursts with Severe Adverse Events in Children. JAMA Pediatrics 2021;175:723-729.

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