Gastroenterology

Gastroenterology

Volume 122, Issue 3, March 2002, Pages 800-805
Gastroenterology

Case Reports
Successful infliximab treatment for steroid-refractory celiac disease: A case report,☆☆

https://doi.org/10.1053/gast.2002.31874Get rights and content

Abstract

Celiac disease is a T cell–mediated enteropathy induced by gluten in genetically predisposed individuals. The majority of patients responds to a gluten-free diet but a small number do not. After the exclusion of gluten in the diet, ulcerative jejunititis, and an enteropathy-associated T-cell lymphoma, another treatment modalities, such as systemic steroids and immunosuppressives, may be necessary. This article reports the case of a 47-year-old white woman with immunoglobulin A deficiency. She was diagnosed with celiac disease with subtotal villous atrophy on jejunal biopsy together with positive antiendomysium and antigliadin immunoglobulin G antibodies. Despite close adherence to a gluten-free diet, her weight continued to decrease, she had diarrhea, and her distal duodenal histology showed no improvement. Some improvement in her symptoms was observed with cyclosporine and systemic steroids, but this was not sustained. Recent evidence has suggested that anti–tumor necrosis factor α antibodies have a role in the amelioration of an animal model of villous atrophy, and after careful consideration, she was treated with infliximab. There was a dramatic improvement in her weight, symptoms, and distal duodenal histology. The response has been maintained for 18 months while on azathioprine therapy. It is concluded that infliximab is an effective treatment that may be considered in a small number of patients with refractory celiac disease, resistant to other therapy.

GASTROENTEROLOGY 2002;122:800-805

Section snippets

Case report

A 47-year-old woman from the north of Scotland presented in December 1993 to her local hospital with a 5-year history of profuse intractable diarrhea and weight loss. Initial investigations included a normal barium follow through, colonoscopy and biopsies, glucose hydrogen breath test, and computerized tomography scan of abdomen. In addition, antibody to double-stranded DNA was negative (<10 U). In February 1995, a jejunal biopsy showed subtotal villous atrophy and an increased intraepithelial

Discussion

We describe a case of a white female with a diagnosis of celiac disease confirmed by elevated titers of antigliadin and antiendomysium IgG antibodies together with subtotal villous atrophy in jejunal and distal duodenal histology. She was IgA deficient. Her disease was uncontrolled despite good compliance with a gluten-free diet. Further treatment with immunosuppressives and oral steroids did not result in a sufficient clinical response, and she continued to complain of diarrhea and weight loss

Acknowledgements

The authors thank John Bode, Edinburgh, Scotland, for help in histopathology and J. A. Garrote and E. Arranz, Universidad de Valladolid, Spain, and the Scottish Blood Transfusion Service, Edinburgh, Scotland, for help in DQ-2 typing.

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  • Cited by (0)

    Address requests for reprints to: Subrata Ghosh, M.D., F.R.C.P., Gastrointestinal Laboratory, Western General Hospital, Crewe Road, Edinburgh, EH4 2XU, Scotland. e-mail: [email protected]; fax: (44) 131-537-1007.

    ☆☆

    Drs. Gillett and Arnott made equal contributions to this article.

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