Case Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer

Introduction: Autoimmune pancreatitis (AIP) is a rare extraintestinal manifestation of inflammatory bowel disease (IBD) which is typically responsive to corticosteroid treatment.Case Presentation: We report a case of a 17-year-old male diagnosed with ulcerative colitis who subsequently developed acu...

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Autores principales: Sujin Choi, Hae Jeong Lee, An Na Seo, Han Ik Bae, Hyung Jun Kwon, Chang Min Cho, So Mi Lee, Byung-Ho Choe, Ben Kang
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Publicado: Frontiers Media S.A. 2021
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spelling oai:doaj.org-article:52d90b34e6ab4ff2ad5d07afdaebf6612021-12-01T08:04:10ZCase Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer2296-236010.3389/fped.2021.791840https://doaj.org/article/52d90b34e6ab4ff2ad5d07afdaebf6612021-11-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fped.2021.791840/fullhttps://doaj.org/toc/2296-2360Introduction: Autoimmune pancreatitis (AIP) is a rare extraintestinal manifestation of inflammatory bowel disease (IBD) which is typically responsive to corticosteroid treatment.Case Presentation: We report a case of a 17-year-old male diagnosed with ulcerative colitis who subsequently developed acute pancreatitis. Blood tests demonstrated elevated pancreatic enzyme levels of amylase (1319 U/L) and lipase (809 U/L). Abdominal computed tomography revealed peripancreatic fat stranding and the presence of a perisplenic pseudocyst. Azathioprine and mesalazine were stopped as possible causes of drug-induced pancreatitis. However, pancreatic enzymes remained elevated and corticosteroid treatment was started. Despite corticosteroid therapy, amylase and lipase levels continued to increase. Infliximab was started due to a flare in gastrointestinal symptoms of ulcerative colitis. Follow-up abdominal ultrasonography revealed a pancreatic tail mass. Tumor markers, including CA 19-9, were elevated and atypical cells were seen on histological examination of an endoscopic ultrasonography-guided fine needle aspiration biopsy. Surgical pancreaticosplenectomy was performed for suspected pancreatic neoplasm. Surprisingly, histology revealed chronic pancreatitis with storiform fibrosis and infiltration of IgG4-positive cells, compatible with AIP type 1. Thereafter, pancreatic enzymes gradually decreased to normal levels and the patient has been in remission for 9 months on infliximab monotherapy.Conclusion: Pediatric gastroenterologists should keep in mind that AIP may develop during the natural course of pediatric IBD. Moreover, the development of pancreatic fibrosis may be non-responsive to corticosteroid treatment and mimic pancreatic neoplasia.Sujin ChoiHae Jeong LeeAn Na SeoHan Ik BaeHyung Jun KwonChang Min ChoSo Mi LeeByung-Ho ChoeBen KangFrontiers Media S.A.articleautoimmune pancreatitisulcerative colitisinflammatory bowel diseasepancreatic cancerIgG4PediatricsRJ1-570ENFrontiers in Pediatrics, Vol 9 (2021)
institution DOAJ
collection DOAJ
language EN
topic autoimmune pancreatitis
ulcerative colitis
inflammatory bowel disease
pancreatic cancer
IgG4
Pediatrics
RJ1-570
spellingShingle autoimmune pancreatitis
ulcerative colitis
inflammatory bowel disease
pancreatic cancer
IgG4
Pediatrics
RJ1-570
Sujin Choi
Hae Jeong Lee
An Na Seo
Han Ik Bae
Hyung Jun Kwon
Chang Min Cho
So Mi Lee
Byung-Ho Choe
Ben Kang
Case Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer
description Introduction: Autoimmune pancreatitis (AIP) is a rare extraintestinal manifestation of inflammatory bowel disease (IBD) which is typically responsive to corticosteroid treatment.Case Presentation: We report a case of a 17-year-old male diagnosed with ulcerative colitis who subsequently developed acute pancreatitis. Blood tests demonstrated elevated pancreatic enzyme levels of amylase (1319 U/L) and lipase (809 U/L). Abdominal computed tomography revealed peripancreatic fat stranding and the presence of a perisplenic pseudocyst. Azathioprine and mesalazine were stopped as possible causes of drug-induced pancreatitis. However, pancreatic enzymes remained elevated and corticosteroid treatment was started. Despite corticosteroid therapy, amylase and lipase levels continued to increase. Infliximab was started due to a flare in gastrointestinal symptoms of ulcerative colitis. Follow-up abdominal ultrasonography revealed a pancreatic tail mass. Tumor markers, including CA 19-9, were elevated and atypical cells were seen on histological examination of an endoscopic ultrasonography-guided fine needle aspiration biopsy. Surgical pancreaticosplenectomy was performed for suspected pancreatic neoplasm. Surprisingly, histology revealed chronic pancreatitis with storiform fibrosis and infiltration of IgG4-positive cells, compatible with AIP type 1. Thereafter, pancreatic enzymes gradually decreased to normal levels and the patient has been in remission for 9 months on infliximab monotherapy.Conclusion: Pediatric gastroenterologists should keep in mind that AIP may develop during the natural course of pediatric IBD. Moreover, the development of pancreatic fibrosis may be non-responsive to corticosteroid treatment and mimic pancreatic neoplasia.
format article
author Sujin Choi
Hae Jeong Lee
An Na Seo
Han Ik Bae
Hyung Jun Kwon
Chang Min Cho
So Mi Lee
Byung-Ho Choe
Ben Kang
author_facet Sujin Choi
Hae Jeong Lee
An Na Seo
Han Ik Bae
Hyung Jun Kwon
Chang Min Cho
So Mi Lee
Byung-Ho Choe
Ben Kang
author_sort Sujin Choi
title Case Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer
title_short Case Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer
title_full Case Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer
title_fullStr Case Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer
title_full_unstemmed Case Report: Development of Type 1 Autoimmune Pancreatitis in an Adolescent With Ulcerative Colitis Mimicking Pancreatic Cancer
title_sort case report: development of type 1 autoimmune pancreatitis in an adolescent with ulcerative colitis mimicking pancreatic cancer
publisher Frontiers Media S.A.
publishDate 2021
url https://doaj.org/article/52d90b34e6ab4ff2ad5d07afdaebf661
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