Immunoglobulin G4‐related sclerosing oesophagitis in a 9‐year‐old girl

Bienfait, Lucie; Nagy, Nathalie; Moussaoui, Imad El; Lingier, Pierre; Nakadi, Issam El; Demetter, Pieter; Verset, Laurine

doi:10.1111/his.13479

Cited by 3 publications

(9 citation statements)

References 5 publications

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“…43 IgG4-related oesophagitis is exceedingly rare in the literature, with a handful of case reports and one case-series to date. [44][45][46][47][48][49][50][51][52][53] Awareness and recognition of IgG4-related disease is important due to its propensity to be misdiagnosed as malignancy, based on its clinical and radiological appearance. 44 IgG4-related oesophagitis should be considered in unexplained strictures of the oesophagus, as treatment with steroids may prevent unnecessary oesophagectomies.…”

Section: Igg4-related Oesophagitismentioning

confidence: 99%

“…Symptom duration ranged from a few months to 20 years (median = 3 years) prior to establishing a diagnosis of IgG4-related oesophagitis. 51,52…”

Section: L I N I C a L F E A T U R E Smentioning

confidence: 99%

“…The case reports provide similar findings to this study, demonstrating 10-200 IgG4 + plasma cells/HPF (median >50) and an IgG4:IgG ratio ranging from 30 to >90% (median = 60%). [44][45][46][47][48][49][50]52,53…”

Section: H I S T O P a T H O L O G Ymentioning

confidence: 99%

“…The duration of symptoms is highly variable, some of which may be attributed to delayed diagnosis of this rare entity. Symptom duration ranged from a few months to 20 years (median = 3 years) prior to establishing a diagnosis of IgG4‐related oesophagitis 51,52 …”

Section: Igg4‐related Oesophagitismentioning

confidence: 99%

“…Involvement of the biliary tract, lymph nodes, salivary glands, kidneys, thyroid and skin has also been described 43 . IgG4‐related oesophagitis is exceedingly rare in the literature, with a handful of case reports and one case‐series to date 44–53 . Awareness and recognition of IgG4‐related disease is important due to its propensity to be misdiagnosed as malignancy, based on its clinical and radiological appearance 44 .…”

Section: Igg4‐related Oesophagitismentioning

confidence: 99%

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Novel and rare forms of oesophagitis

Malone

Sheahan

2020

Histopathology

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Our understanding of inflammatory diseases of the gastrointestinal tract, including those of the oesophagus, has expanded in recent years. Once attributed almost exclusively to gastro‐oesophageal reflux disease or infection, it is now recognised that oesophagitis may occur due to a variety of distinct disease entities. Many of these conditions cause debilitating and persistent symptoms, impacting upon quality of life and necessitating ongoing surveillance and treatment. This review will consider the clinical, endoscopic and histopathological features of these novel and rare forms of oesophagitis.

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Section: Igg4-related Oesophagitismentioning

confidence: 99%

“…Symptom duration ranged from a few months to 20 years (median = 3 years) prior to establishing a diagnosis of IgG4-related oesophagitis. 51,52…”

Section: L I N I C a L F E A T U R E Smentioning

confidence: 99%

Section: H I S T O P a T H O L O G Ymentioning

confidence: 99%

Section: Igg4‐related Oesophagitismentioning

confidence: 99%

Section: Igg4‐related Oesophagitismentioning

confidence: 99%

See 3 more Smart Citations

Novel and rare forms of oesophagitis

Malone

Sheahan

2020

Histopathology

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Esophagitis in patients without gastroesophageal reflux disease or eosinophilic esophagitis

Lisovsky

Srivastava²

2019

Current Opinion in Gastroenterology

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Frequency, clinical picture, diagnosis, and differential diagnosis of the pathology of the digestive tract (esophagus, stomach, and bowel) in IgG4-related disease (autoimmune pancreatitis)

Gubergrits

2023

Bul. of the Club of Pancr.

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Despite the publication of European guidelines for the diagnosis and treatment of IgG4-related disease (IgG4-RD), this problem is very complicated from a practical point of view. There are many publications about autoimmune pancreatitis and liver and biliary tract lesions in IgG4-RD, but the data on the digestive tract lesions are not sufficient. Gastrointestinal manifestations of IgG4-RD are considered to be rare or very rare. IgG4-RD presenting as esophageal lesions is rare condition. Only single clinical cases are available. I. Obiorah et al. reported the highest number of cases of IgG4-related esophagitis (8 cases). Interestingly, 7/8 cases reported by I. Obiorah et al. were males (age range: 18 to 79 years), and most patients had dysphagia as the most prominent symptom, with erosions and strictures at endoscopy. Histologically, all patients had a lymphoplasmacytic infiltrate and storiform fibrosis, while obliterative phlebitis was observed in 3/8 patients. Hence, 2 of the 3 major histological “Boston criteria” of IgG4-RD were fulfilled in all patients. Besides, all patients had at least 50 IgG4-positive cells per HPF (range: 50–110), and IgG4/IgG ratios of at least 50% (range: 50–90%). Of I. Obiorah’s patients, s-IgG4 had only been measured in three — all of whom had values in the normal range. Esophageal strictures were identified in three patients, two patients had achalasia. Based on K. Notohara’s series and the other published cases, it can be concluded that gastric IgG4-RD often (but not always) seems to be clinically asymptomatic, that s-IgG4 is elevated in a considerable number of cases, and histologically, that at least two of the Boston criteria often are found. The recommended cut-off of 50 IgG4-positive cells and IgG4/IgG ratios of >40% was often fulfilled in the reported cases of IgG4-RD. There are few reports of IgG4-RD involving the duodenum, duodenal papilla, ileum with small bowel obstruction caused by pseudotumor or sclerosing disease of the small bowel with luminal narrowing. There are case reports of IgG4-RD with lesion of ileocecal area with an obstructing ileocecal mass and appendix, mimicking Crohn’s disease, appendicitis or appendiceal tumor. IgG4-related inflammatory bowel disease may — rarely — also be one of manifestations of IgG4-RD. Treatment should be conducted in accordance with European guidelines.

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Immunoglobulin G4‐related sclerosing oesophagitis in a 9‐year‐old girl

Cited by 3 publications

References 5 publications

Novel and rare forms of oesophagitis

Novel and rare forms of oesophagitis

Esophagitis in patients without gastroesophageal reflux disease or eosinophilic esophagitis

Frequency, clinical picture, diagnosis, and differential diagnosis of the pathology of the digestive tract (esophagus, stomach, and bowel) in IgG4-related disease (autoimmune pancreatitis)

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