Original Article

Excess primary intestinal lymphoproliferative disorders in patients with inflammatory bowel disease†

  1. Harry Sokol MD1,
  2. Laurent Beaugerie MD1,*,
  3. Marc Maynadié MD2,
  4. David Laharie MD3,
  5. Jean-Louis Dupas MD4,
  6. Bernard Flourié MD5,
  7. Eric Lerebours MD6,
  8. Laurent Peyrin-Biroulet MD7,
  9. Matthieu Allez MD8,
  10. Tabassome Simon MD9,
  11. Fabrice Carrat MD10,
  12. Nicole Brousse MD11,
  13. for the CESAME Study Group1

Article first published online: 23 JAN 2012

DOI: 10.1002/ibd.22889

Issue

Inflammatory Bowel Diseases

Inflammatory Bowel Diseases

Volume 18, Issue 11, pages 2063–2071, November 2012

Additional Information

How to Cite

Sokol, H., Beaugerie, L., Maynadié, M., Laharie, D., Dupas, J.-L., Flourié, B., Lerebours, E., Peyrin-Biroulet, L., Allez, M., Simon, T., Carrat, F., Brousse, N. and for the CESAME Study Group (2012), Excess primary intestinal lymphoproliferative disorders in patients with inflammatory bowel disease. Inflamm Bowel Dis, 18: 2063–2071. doi: 10.1002/ibd.22889

Author Information

  1. 1

    Department of Gastroenterology, AP-HP, Hôpital Saint-Antoine and UPMC University of Paris, Paris, France

  2. 2

    Registry of Hematological Malignancies of Côte d'Or, University of Burgundy and Hematology laboratory, CHU of Dijon, France

  3. 3

    Department of Gastroenterology, Hôpital Haut-Lévêque, CHU de Bordeaux, Pessac, France

  4. 4

    Department of Gastroenterology, Amiens University Hospital, Amiens, France

  5. 5

    Department of Gastroenterology, Centre Hospitalier Lyon Sud, Pierre Benite, France

  6. 6

    Department of Gastroenterology. Rouen University Hospital, Rouen, France

  7. 7

    Department of Gastroenterology and INSERM U954, Nancy University Hospital, Henri Poincaré University, Nancy 1, Vandœuvre-lès-Nancy, France

  8. 8

    Department of Gastroenterology, AP-HP, Hôpital Saint-Louis and Université Denis Diderot Paris 7, Paris, France

  9. 9

    Department of Pharmacology, AP-HP, Hôpital Saint-Antoine, URCEST, and UPMC University of Paris, Paris, France

  10. 10

    Unit of Public Health, AP-HP, Hôpital Saint-Antoine, INSERM UMR-S 707, and UMR-S 707 UPMC University of Paris, Paris, France

  11. 11

    Department of Pathology, AP-HP, Hôpital Necker-Enfants Malades, Université Paris Descartes Paris-V, Paris, France

*Service de Gastroentérologie et Nutrition, Hôpital Saint-Antoine, 184 rue du faubourg Saint-Antoine, 75571 Paris Cedex 12, France

  1. †

    Supported by grants from Programme Hospitalier de Recherche Clinique National (AOM05157), Association François Aupetit, Délégation Inter-régionale de la Recherche Clinique Ile de France-Assistance Publique Hôpitaux de Paris (AP-HP), Ligue contre le Cancer, Fonds de Recherche de la Société Nationale Française de Gastro-entérologie, and Institut de Recherche sur les Maladies de l'Appareil Digestif (IRMAD).

Publication History

  1. Issue published online: 15 OCT 2012
  2. Article first published online: 23 JAN 2012
  3. Manuscript Accepted: 22 DEC 2011
  4. Manuscript Received: 16 DEC 2011

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Keywords:

  • CESAME study;
  • primary intestinal lymphomas;
  • inflammatory bowel disease;
  • thiopurines

Abstract

Background:

It remains to be shown whether inflammatory bowel disease (IBD) is associated with an increased risk of primary intestinal lymphoproliferative disorders (PILD). We assessed this risk in the CESAME French nationwide prospective observational cohort.

Methods:

In all, 680 gastroenterologists enrolled 19,486 patients with IBD (Crohn's disease in 60.3%) from May 2004 to June 2005. Follow-up ended on 31 December 2007. Available biopsy samples and surgical specimens from patients with PILD (n = 14) were centralized for review. The reference incidence of PILD in the general population was obtained from the Côte d'Or registry and was used as a comparator to assess the standardized incidence ratio (SIR). The influence of thiopurine exposure was explored in a nested case-control study.

Results:

In the CESAME population the crude incidence of PILD was 0.12/1000 patient-years, with a corresponding SIR of 17.51 (95% confidence interval [CI], 6.43–38.11; P < 0.0001). The risk was highest in patients exposed to thiopurines (SIR 49.52, 95% CI 13.49–126.8; P < 0.0001), while it did not reach statistical significance in patients naïve to thiopurines (SIR 4.83, 95% CI, 0.12–26.91; P = 0.37). The odds ratio associated with ongoing thiopurine exposure (vs. naïve) was 2.97 (95% CI, 0.30–infinity; P = 0.38). All 14 cases of PILD were non-Hodgkin's B-cell LD, 78.6% occurred in males, 85.7% arose in IBD lesions, and 45.5% were Epstein–Barr virus-positive. Eleven cases occurred in patients with Crohn's disease. Mean (SD) age at PILD diagnosis was 55.1 (5.6) years and the median time since IBD onset was 8.0 years (interquartile range, 3.0–15.8).

Conclusions:

Patients with IBD have an increased risk of developing PILD. (Inflamm Bowel Dis 2012;)

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