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A severe but reversible refractory sprue
  1. X Dray1,
  2. F Joly2,
  3. A Lavergne-Slove3,
  4. X Treton4,
  5. Y Bouhnik4,
  6. B Messing4
  1. 1Département de Pathologie Digestive, Hôpital Lariboisière, Paris, France
  2. 2Service d’Hépato-gastroentérologie et d’Assistance Nutritive, Hôpital Beaujon, Clichy la Garenne, France
  3. 3Service Central d’Anatomie et Cytologie Pathologiques, Hôpital Lariboisière, Paris, France
  4. 4Service d’Hépato-gastroentérologie et d’Assistance Nutritive, Hôpital Beaujon, Clichy la Garenne, France
  1. Correspondence to:
    Dr X Dray
    Département de Pathologie Digestive, Hôpital Lariboisière, 2 rue Ambroise Paré, 75475 Paris cedex, France; xavier.dray{at}lrb.aphp.fr

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Original treatments, including corticosteroids, azathioprine, infliximab, ciclosporin, and interleukin 10 have been occasionally reported in refractory sprue patients with unsatisfactory results.1–8 We report on the clinical and histological efficacy of anti-T chemotherapy (pentostatine) in a patient with a refractory sprue complicated by an ulcerative and stenotic jejunitis.

A 38 year old man diagnosed with refractory sprue in June 2002 developed ulcerative jejunitis in July 2003. Diagnosis led on the following: malabsorptive diarrhoea resistant to a gluten free diet (GFD); severe malnutrition (body mass index (BMI) 14.3 kg/m2); albuminaemia 11 g/l; diffuse total villous atrophy associated with jejunal ulcerations and a major intestinal intraepithelial CD3+ CD8− T lymphocyte population (IEL) (fig 1A–C); clonal T cell receptor γ rearrangement; negative circulating antigliadin and antitransglutaminase IgA and IgG antibodies (total IgA and IgG were normal); hyposplenism; …

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