IgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girl

Abstract Background Immunoglobulin G4-related disease (IgG4-RD) is a systemic inflammatory disease and affected individuals typically present with an increased infiltration of IgG4-positive plasma cells in the pancreas, hepatobiliary tract, and liver but rarely in the gastrointestinal tract. Case pr...

Full description

Bibliographic Details
Main Authors: Daiki Kato, Hiroo Uchida, Akinari Hinoki, Wataru Sumida, Chiyoe Shirota, Satoshi Makita, Masamune Okamoto, Aitaro Takimoto, Shunya Takada, Yoichi Nakagawa
Format: Article
Language:English
Published: BMC 2023-07-01
Series:BMC Pediatrics
Subjects:
Online Access:https://doi.org/10.1186/s12887-023-04190-z
_version_ 1797769335090970624
author Daiki Kato
Hiroo Uchida
Akinari Hinoki
Wataru Sumida
Chiyoe Shirota
Satoshi Makita
Masamune Okamoto
Aitaro Takimoto
Shunya Takada
Yoichi Nakagawa
author_facet Daiki Kato
Hiroo Uchida
Akinari Hinoki
Wataru Sumida
Chiyoe Shirota
Satoshi Makita
Masamune Okamoto
Aitaro Takimoto
Shunya Takada
Yoichi Nakagawa
author_sort Daiki Kato
collection DOAJ
description Abstract Background Immunoglobulin G4-related disease (IgG4-RD) is a systemic inflammatory disease and affected individuals typically present with an increased infiltration of IgG4-positive plasma cells in the pancreas, hepatobiliary tract, and liver but rarely in the gastrointestinal tract. Case presentation A 12-year-old girl presented with vomiting and poor weight gain. Gastroscopy revealed duodenal stenosis and ulceration. Computed tomography revealed edematous duodenal wall thickening and air-fluid levels on the right side of the duodenum, which suggested duodenal perforation or penetration. She underwent pancreaticoduodenectomy, and IgG4-RD was diagnosed via histopathology. Conclusions This is the first pediatric case of isolated duodenal IgG4-RD resulting in duodenal obstruction after multiple ulcers. Gastrointestinal IgG4-RD should be among the differential diagnoses of unexplained gastrointestinal obstruction or ulceration even in children.
first_indexed 2024-03-12T21:07:31Z
format Article
id doaj.art-e6a4c7787bc948cca7a8bb72faec352f
institution Directory Open Access Journal
issn 1471-2431
language English
last_indexed 2024-03-12T21:07:31Z
publishDate 2023-07-01
publisher BMC
record_format Article
series BMC Pediatrics
spelling doaj.art-e6a4c7787bc948cca7a8bb72faec352f2023-07-30T11:24:54ZengBMCBMC Pediatrics1471-24312023-07-012311410.1186/s12887-023-04190-zIgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girlDaiki Kato0Hiroo Uchida1Akinari Hinoki2Wataru Sumida3Chiyoe Shirota4Satoshi Makita5Masamune Okamoto6Aitaro Takimoto7Shunya Takada8Yoichi Nakagawa9Department of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Rare/Intractable Cancer Analysis Research, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineDepartment of Pediatric Surgery, Nagoya University Graduate School of MedicineAbstract Background Immunoglobulin G4-related disease (IgG4-RD) is a systemic inflammatory disease and affected individuals typically present with an increased infiltration of IgG4-positive plasma cells in the pancreas, hepatobiliary tract, and liver but rarely in the gastrointestinal tract. Case presentation A 12-year-old girl presented with vomiting and poor weight gain. Gastroscopy revealed duodenal stenosis and ulceration. Computed tomography revealed edematous duodenal wall thickening and air-fluid levels on the right side of the duodenum, which suggested duodenal perforation or penetration. She underwent pancreaticoduodenectomy, and IgG4-RD was diagnosed via histopathology. Conclusions This is the first pediatric case of isolated duodenal IgG4-RD resulting in duodenal obstruction after multiple ulcers. Gastrointestinal IgG4-RD should be among the differential diagnoses of unexplained gastrointestinal obstruction or ulceration even in children.https://doi.org/10.1186/s12887-023-04190-zIgG4-related diseaseGastrointestinal ulcerationGastric outlet obstruction
spellingShingle Daiki Kato
Hiroo Uchida
Akinari Hinoki
Wataru Sumida
Chiyoe Shirota
Satoshi Makita
Masamune Okamoto
Aitaro Takimoto
Shunya Takada
Yoichi Nakagawa
IgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girl
BMC Pediatrics
IgG4-related disease
Gastrointestinal ulceration
Gastric outlet obstruction
title IgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girl
title_full IgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girl
title_fullStr IgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girl
title_full_unstemmed IgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girl
title_short IgG4-related disease of duodenal obstruction due to multiple ulcers in a 12-year-old girl
title_sort igg4 related disease of duodenal obstruction due to multiple ulcers in a 12 year old girl
topic IgG4-related disease
Gastrointestinal ulceration
Gastric outlet obstruction
url https://doi.org/10.1186/s12887-023-04190-z
work_keys_str_mv AT daikikato igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT hiroouchida igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT akinarihinoki igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT watarusumida igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT chiyoeshirota igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT satoshimakita igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT masamuneokamoto igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT aitarotakimoto igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT shunyatakada igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl
AT yoichinakagawa igg4relateddiseaseofduodenalobstructionduetomultipleulcersina12yearoldgirl