Abstract
A 40-year-old man presented with insidious onset dysphagia for both solids and liquids for 4 years with recurrent oral ulcerations. On examination he was anemic, and barium swallow demonstrated a web in the postcricoid region. As part of the workup for unexplained iron deficiency anemia, a duodenal biopsy was taken that revealed moderate flattening of villi with increased intraepithelial lymphocytes consistent with the diagnosis of celiac disease. However, the serological tests for celiac disease (IgA antiendomysial antibody, IgA antitissue transglutaminase antibody, and IgA antigliadin antibody) were all negative. Serum level of IgA was markedly low. A diagnosis of atypical celiac disease with severe selective IgA deficiency was made. After the institution of a gluten-free diet (GFD), his general condition as well as anemia improved. Histological recovery was documented on repeat duodenal biopsy 6 months after GFD.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
References
AS Puri S Garg R Monga et al. (2004) ArticleTitleSpectrum of atypical celiac disease in North Indian children Ind Pediatr 41 822–7
PD Burrows MD Cooper (1997) ArticleTitleIgA deficiency Adv Immunol 65 245 Occurrence Handle1:CAS:528:DyaK2sXntVehsrc%3D Occurrence Handle9238511
HE Prince GL Lorman WL BInder (2000) ArticleTitleImmunoglobulin A (IgA) and alternative celiac disease-associated antibodies in sera submitted to a reference laboratory for endomysial IgA testing Clin Diagn Lab Immunol 7 192–6 Occurrence Handle1:CAS:528:DC%2BD3cXitVCqtLc%3D Occurrence Handle10702491 Occurrence Handle10.1128/CDLI.7.2.192-196.2000
V Kumar MJ Chorzelska J Sulej K Karnewska T Farrel S Jablonska (2002) ArticleTitleCeliac disease and immunoglobulin A deficiency: how effective are the serological methods of diagnosis Clin Diagn Lab Immunol 9 1295–300 Occurrence Handle1:CAS:528:DC%2BD38XptFakt7o%3D Occurrence Handle12414763
M Chisolm GM Ardran ST Callender et al. (1971) ArticleTitleA follow-up study of patients with postcricoid webs Q J Med 159 409–20
A Jacobs GS Kilpatrick (1964) ArticleTitleThe Paterson–Kelly syndrome Br Med J 2 79–82
JK Bredenkamp DJ Castro RA Mickel (1990) ArticleTitleImportance of iron repletion in the management of Plummer–Vinson syndrome Ann Otol Rhinol Laryngol 99 51–4 Occurrence Handle1:STN:280:By%2BC3czjsVE%3D Occurrence Handle2294834
M Chisolm (1974) ArticleTitleThe association between webs, iron and postcricoid carcinoma Postgrad Med J 50 215–19
Z Rashid A Kumar M Komar (1999) ArticleTitlePlummer–Vinson syndrome and postcricoid carcinoma: late complications of unrecognized celiac disease Am J Gastroenterol 94 1991 Occurrence Handle1:STN:280:DyaK1Mzjt12hsA%3D%3D Occurrence Handle10406289 Occurrence Handle10.1111/j.1572-0241.1999.01991.x
W Jessner H Vogelsang A Puspok P Ferenci A Gangl G Novacek et al. (2003) ArticleTitlePlummer–Vinson syndrome associated with celiac disease and complicated by postcricoid carcinoma and carcinoma of the tongue Am J Gastroenterol 98 1208–9 Occurrence Handle12809857 Occurrence Handle10.1111/j.1572-0241.2003.07438.x
W Dickey B McConnell (1999) ArticleTitleCeliac disease presenting as the Paterson–Brown–Kelly (Plummer–Vinson) syndrome Am J Gastroenterol 94 527–9 Occurrence Handle1:STN:280:DyaK1M7ksFalsg%3D%3D Occurrence Handle10022662
P Malhotra R Kochar N Varma S Kumari S Jain S Varma (2000) ArticleTitlePaterson–Kelly syndrome and celiac disease: a rare combination Ind J Gastroenterol 19 191–2 Occurrence Handle1:STN:280:DC%2BD3crgvVCqsA%3D%3D
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Saroj, D., Naresh, A. & Puri, A. Atypical celiac disease with IgA deficiency presenting as Plummer–Vinson syndrome: a case report. Esophagus 3, 23–25 (2006). https://doi.org/10.1007/s10388-006-0069-5
Received:
Accepted:
Issue Date:
DOI: https://doi.org/10.1007/s10388-006-0069-5