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 Table of Contents  
IMAGES AND VIDEOS
Year : 2016  |  Volume : 5  |  Issue : 6  |  Page : 393-395

An unusual cause of dysphagia: Isolated IgG 4-related esophageal disease


1 Department of Histopathology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
2 Department of Gastroenterology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
3 Department of Nuclear Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India
4 Department of Surgery, Postgraduate Institute of Medical Education and Research, Chandigarh, India

Date of Submission18-Oct-2015
Date of Acceptance22-Apr-2016
Date of Web Publication15-Dec-2016

Correspondence Address:
Dr. Surinder Singh Rana
Department of Gastroenterology, Postgraduate Institute of Medical Education and Research, Chandigarh - 160 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2303-9027.195872

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How to cite this article:
Nada R, Rana SS, Sharma R, Dhalaria L, Singh H, Mittal BR, Gupta R, Bhasin DK. An unusual cause of dysphagia: Isolated IgG 4-related esophageal disease. Endosc Ultrasound 2016;5:393-5

How to cite this URL:
Nada R, Rana SS, Sharma R, Dhalaria L, Singh H, Mittal BR, Gupta R, Bhasin DK. An unusual cause of dysphagia: Isolated IgG 4-related esophageal disease. Endosc Ultrasound [serial online] 2016 [cited 2022 Jan 27];5:393-5. Available from: http://www.eusjournal.com/text.asp?2016/5/6/393/195872

A 60-year-male presented with progressively increasing dysphagia accompanied with loss of weight. He denied ingestion of corrosive or any medication. Upper gastrointestinal endoscopy revealed a nonnegotiable stricture at 32 cm from the incisors [Figure 1] and multiple endoscopic biopsies from the mouth of the stricture were noncontributory. Barium swallow confirmed the presence of lower esophageal stricture [Figure 2]. Contrast enhanced computed tomography revealed marked wall thickening of the esophagus at the site of narrowing [Figure 3]. High-frequency endoscopic ultrasound (EUS) at 20 MHz revealed marked thickening of the wall of the esophagus with the loss of wall stratification (maximum thickness 11.2 mm) [Figure 4]. No significant mediastinal lymph nodes were detected on EUS. Endoscopic bougie dilatation of the esophageal stricture was done, and the patient had transient improvement in dysphagia. Positron emission tomography revealed flourodeoxyglucose (FDG) avid esophageal wall thickening (SUVmax 2.3) [Figure 5]. No significant FDG uptake was observed anywhere else in the body. Because of the possibility of hidden malignancy, the patient underwent esophagectomy with gastric pull-up. The resected specimen revealed ulcerated esophageal mucosa and dense submucosal fibro-inflammatory reaction which had storiform pattern and IgG4 rich plasma cell infiltration [Figure 6] and [Figure 7]. Serum IgG4 levels done postoperatively were within normal limits. The patient had an anastomotic leak that was successfully managed with endoscopic dilatation and patient is asymptomatic at 8 months of follow-up.
Figure 1. Upper gastrointestinal endoscopy: Nonnegotiable stricture in lower esophagus

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Figure 2. Barium swallow: Lower esophageal stricture

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Figure 3. Contrast enhanced computed tomography: Marked wall thickening of the esophagus at the site of narrowing

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Figure 4. High-frequency endoscopic ultrasound: Marked thickening of the wall of the esophagus with loss of wall stratification

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Figure 5. Positron emission tomography-computed tomography: Flourodeoxyglucose avid esophageal wall thickening (SUVmax 2.3)

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Figure 6. Microphotograph: Fibro-inflammatory reaction with storiform pattern and plasma rich infiltrate (H and E, ×40)

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Figure 7. Immunohistochemistry: IgG4 rich plasma cells (×40)

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The IgG4 related disease usually involves pancreas with the involvement of biliary tract, salivary glands, lymph nodes, thyroid, kidneys, lung, skin, prostate, and aorta also reported in the literature.[1],[2] Involvement of upper gastrointestinal tract by IgG4-related inflammatory cells is very rare and there are very few case reports describing IgG4-related esophageal disease with the majority of patients being treated with surgery.[2],[3],[4] In spite of rarity, IgG4 related esophageal disease should be considered in the differential diagnosis of unexplained esophageal strictures.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Stone JH, Zen Y, Deshpande V. IgG4-related disease. N Engl J Med 2012;366:539-51.  Back to cited text no. 1
    
2.
Dumas-Campagna M, Bouchard S, Soucy G, et al. IgG4-related esophageal disease presenting as esophagitis dissecans superficialis with chronic strictures. J Clin Med Res 2014;6:295-8.  Back to cited text no. 2
    
3.
Lee H, Joo M, Song TJ, et al. IgG4-related sclerosing esophagitis: A case report. Gastrointest Endosc 2011;73:834-7.  Back to cited text no. 3
    
4.
Lopes J, Hochwald SN, Lancia N, et al. Autoimmune esophagitis: IgG4-related tumors of the esophagus. J Gastrointest Surg 2010;14:1031-4.  Back to cited text no. 4
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]


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