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

Kato, Daiki; Uchida, Hiroo; Hinoki, Akinari; Sumida, Wataru; Shirota, Chiyoe; Makita, Satoshi; Okamoto, Masayuki; Takimoto, Aitaro; Takada, Shunya; Nakagawa, Yoshifumi

doi:10.1186/s12887-023-04190-z

Cited by 2 publications

(5 citation statements)

References 6 publications

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“… 1 To our knowledge, there are 19 reported cases in the literature that present gastrointestinal obstruction and gastroduodenal ulcer. 1 , 4 , 5 , 6 Among these 19 cases, there was only one case of multiple duodenal ulcers leading to duodenal obstruction. 5 Although these ulcers have been described as IgG4‐RD, no specific relationship with type 1 AIP has been noted.…”

Section: Discussionmentioning

confidence: 99%

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Immunoglobulin G4 ‐related gastrointestinal disease associated with type 1 autoimmune pancreatitis: A case report

Sato,

Kasai,

Eto

et al. 2024

DEN Open

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Immunoglobulin G4 (IgG4)‐related diseaseis a systemic inflammatory condition of unknown etiology characterized by increases in serum IgG4 and in the number of IgG4‐positive cells in affected tissues. One of the commonly involved locations is the pancreas; this condition is known as type 1 autoimmune pancreatitis (AIP). Type 1 AIP, which shows a biliary stricture in the intrapancreatic bile duct, can be misdiagnosed as a malignancy due to similar cholangiography findings and clinical presentation. In rare cases complicated by post‐bulbar duodenal ulcers, differentiating between type 1 AIP and malignancies is even more difficult. An 81‐year‐old male was referred to our hospital for the treatment of a pancreatic head mass and obstructive jaundice. Serological and radiological findings were consistent with both type 1 AIP and a malignancy. Gastroduodenoscopy revealed a post‐bulbar duodenal ulcer with endoscopic features that evoked malignant duodenal invasion. Although biopsies were negative for malignant cells, subsequent bleeding from the lesion suggested the progression of malignancy, which led to surgical resection. Pancreatoduodenectomy and pathological examination indicated that type 1 AIP was present. Simultaneously, the involvement of IgG4‐related disease in the ulcerative lesion was suggested. To our knowledge, this is the first reported case of type 1 AIP complicated by post‐bulbar duodenal ulcers, which was misdiagnosed as malignancy and considered an IgG4‐related gastrointestinal disease associated with type 1 AIP.

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Section: Discussionmentioning

confidence: 99%

“… 1 , 4 , 5 , 6 Among these 19 cases, there was only one case of multiple duodenal ulcers leading to duodenal obstruction. 5 Although these ulcers have been described as IgG4‐RD, no specific relationship with type 1 AIP has been noted.…”

Section: Discussionmentioning

confidence: 99%

Immunoglobulin G4 ‐related gastrointestinal disease associated with type 1 autoimmune pancreatitis: A case report

Sato,

Kasai,

Eto

et al. 2024

DEN Open

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“…Increased serum IgG4 levels and IgG4‐positive plasma levels with lymphoplasmacytic inflammation in the duodenal wall and underlying sclerosing fibrosis on histopathology lead to a subsequent diagnosis of an IgG4‐related duodenal stenosis. 9 …”

Section: Discussionmentioning

confidence: 99%

“…However, the patient returned to the healthcare with complaints of vomiting and poor weight gain, which revealed to be a duodenal stricture on gastroscopy. Increased serum IgG4 levels and IgG4‐positive plasma levels with lymphoplasmacytic inflammation in the duodenal wall and underlying sclerosing fibrosis on histopathology lead to a subsequent diagnosis of an IgG4‐related duodenal stenosis 9 …”

Section: Discussionmentioning

confidence: 99%

“…Increased serum IgG4 levels and IgG4-positive plasma levels with lymphoplasmacytic inflammation in the duodenal wall and underlying sclerosing fibrosis on histopathology lead to a subsequent diagnosis of an IgG4-related duodenal stenosis. 9 Occasionally, an IgG4-RD may recur which occurred in one of the cases where an IgG4-RD patient with mural thickening of the proximal duodenum underwent a Whipple's procedure for resection; however, the patient presented again with abdominal pain, nausea, vomiting, and a raised serum IgG4 level. This was associated with inflammatory changes and duodenal thickening on CT scan while an endoscopy revealed perianastomotic erosions and erythema indicating recurrence of the IgG4-RD.…”

Section: Discussionmentioning

confidence: 99%

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Duodenal stricture secondary to IgG4‐related chronic sclerosing duodenitis—A case report with review of the literature

Anns,

Salar,

Salar

et al. 2024

Clinical Case Reports

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Key Clinical MessageThis case highlights the importance of a definite diagnosis of an IgG4‐related chronic sclerosing duodenitis based on histological and radiological findings to rule out any malignancy in the mass. While dealing with patients having concentric duodenal thickening resulting in stricture formation, one should think of inflammatory etiology as well. IgG4‐related disease is one of these inflammatory disorders where we see soft tissue thickening without a large mass or any associated lymphadenopathy as in our case.AbstractImmunoglobulin G4‐related disease (IgG4‐RD) is distinguished as an infiltration of IgG‐4‐positive plasmacytes involving inflammatory lesions across multiple organs which is accompanied by raised IgG4 levels in the serum. Several inflammatory disorders are recognized as part of the IgG4‐RD family based on shared histopathological features, which include Mikulicz's disease, chronic sclerosing sialadenitis, or Riedel's thyroiditis. Our case highlights a distinctive presentation of IgG4‐related diseases; a 58‐year‐old man presenting with duodenal stricture highly suspicious of a duodenal mass/ampullary mass later found to be due to IgG4‐related sclerosing duodenitis with negative malignancy on biopsy. We present the diagnostic challenges faced and relevant findings noted.

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

Cited by 2 publications

References 6 publications

Immunoglobulin G4 ‐related gastrointestinal disease associated with type 1 autoimmune pancreatitis: A case report

Immunoglobulin G4 ‐related gastrointestinal disease associated with type 1 autoimmune pancreatitis: A case report

Duodenal stricture secondary to IgG4‐related chronic sclerosing duodenitis—A case report with review of the literature

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