Journal of Gastrointestinal Cancer

, Volume 38, Issue 1, pp 19–23 | Cite as

CHOP plus Alemtuzumab can Induce Metabolic Response by FDG-PET but has Minimal Long-term Benefits: A Case Report and Literature Review

  • Sheetal M. Kircher
  • Sandeep Gurbuxani
  • Sonali M. Smith
CASE
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Abstract

Enteropathy-associated T-cell lymphoma (EATL) is a rare subtype of non-Hodgkin’s lymphoma characterized by an aggressive phenotype and poor outcome in the vast majority of cases. Substantial portions of patients have either no prior diagnosis of celiac disease or have a subacute course of celiac disease followed by EATL diagnosis. We report a case of a 72-year-old African American male without history of gastrointestinal intolerance who presented with acute abdominal pain and weight loss leading to the eventual diagnosis of gastric EATL. Despite an initial promising clinical and radiographic response to cyclophosphamide, doxorubicin, vincristine, and prednisone plus alemtuzumab, the disease rapidly progressed with a fatal outcome. This case and review of the literature highlights the features of this uncommon disease and addresses both the diagnostic and therapeutic challenges of this aggressive malignancy. We also discuss our experience with the use of fluoro-2-deoxy-d-glucose positron emission tomography in monitoring treatment response.

Keywords

EATL (enteropathy-associated T-cell lymphoma) alemtuzumab FDG-PET celiac disease therapy 

Notes

References

  1. 1.
    Al-Toma A, Verbeek WH, Hadithi M, et al. Survival in refractory coeliac disease and enteropathy-associated T-cell lymphoma: retrospective evaluation of single-centre experience. Gut. 2007;56:1373–8.PubMedCrossRefGoogle Scholar
  2. 2.
    Alaedini A, Green PH. Narrative review: celiac disease: understanding a complex autoimmune disorder. Ann Intern Med. 2005;142:289–98.PubMedGoogle Scholar
  3. 3.
    Anderson RP, Degano P, Godkin AJ, et al. In vivo antigen challenge in celiac disease identifies a single transglutaminase-modified peptide as the dominant A-gliadin T-cell epitope. Nat Med. 2000;6:337–42.PubMedCrossRefGoogle Scholar
  4. 4.
    Cheson BD, Pfistner B, Juweid ME, et al. Revised response criteria for malignant lymphoma. J Clin Oncol. 2007;25:579–86.PubMedCrossRefGoogle Scholar
  5. 5.
    Daum S, Weiss D, Hummel M, et al. Frequency of clonal intraepithelial T lymphocyte proliferations in enteropathy-type intestinal T cell lymphoma, coeliac disease, and refractory sprue. Gut. 2001;49:804–12.PubMedCrossRefGoogle Scholar
  6. 6.
    Enblad G, Hagberg H, Erlanson M, et al. A pilot study of alemtuzumab (anti-CD52 monoclonal antibody) therapy for patients with relapsed or chemotherapy-refractory peripheral T-cell lymphomas. Blood. 2004;103:2920–4.PubMedCrossRefGoogle Scholar
  7. 7.
    Fasano A, Catassi C. Current approaches to diagnosis and treatment of celiac disease: an evolving spectrum. Gastroenterology. 2001;120:636–51.PubMedCrossRefGoogle Scholar
  8. 8.
    Gale J, Simmonds PD, Mead GM, et al. Enteropathy-type intestinal T-cell lymphoma: clinical features and treatment of 31 patients in a single center. J Clin Oncol. 2000;18:795–803.PubMedGoogle Scholar
  9. 9.
    Gallamini A, Hutchings M, Rigacci L, et al. Early interim 2-[18F]fluoro-2-deoxy-D-glucose positron emission tomography is prognostically superior to international prognostic score in advanced-stage Hodgkin’s lymphoma: a report from a joint Italian–Danish study. J Clin Oncol. 2007;25:3746–52.PubMedCrossRefGoogle Scholar
  10. 10.
    Green PH, Cellier C. Celiac disease. N Engl J Med. 2007;357:1731–43.PubMedCrossRefGoogle Scholar
  11. 11.
    Honemann D, Prince HM, Hicks RJ, et al. Enteropathy-associated T-cell lymphoma without a prior diagnosis of coeliac disease: diagnostic dilemmas and management options. Ann Hematol. 2005;84:118–21.PubMedCrossRefGoogle Scholar
  12. 12.
    Obermann EC, Diss TC, Hamoudi RA, et al. Loss of heterozygosity at chromosome 9p21 is a frequent finding in enteropathy-type T-cell lymphoma. J Pathol. 2004;202:252–62.PubMedCrossRefGoogle Scholar
  13. 13.
    Verbeek WH, Mulder CJ, Zweegman S. Alemtuzumab for refractory celiac disease. N Engl J Med. 2006;355:1396–7. author reply 1397.PubMedCrossRefGoogle Scholar
  14. 14.
    Vivas S, Ruiz de Morales JM, Ramos F, et al. Alemtuzumab for refractory celiac disease in a patient at risk for enteropathy-associated T-cell lymphoma. N Engl J Med. 2006;354:2514–5.PubMedCrossRefGoogle Scholar
  15. 15.
    Wohrer S, Chott A, Drach J, et al. Chemotherapy with cyclophosphamide, doxorubicin, etoposide, vincristine and prednisone (CHOEP) is not effective in patients with enteropathy-type intestinal T-cell lymphoma. Ann Oncol. 2004;15:1680–3.PubMedCrossRefGoogle Scholar

Copyright information

© Humana Press Inc. 2008

Authors and Affiliations

  • Sheetal M. Kircher
    • 1
  • Sandeep Gurbuxani
    • 1
  • Sonali M. Smith
    • 2
  1. 1.Department of Internal MedicineUniversity of Chicago HospitalsChicagoUSA
  2. 2.Section of Hematology/Oncology, Department of MedicineUniversity of Chicago HospitalsChicagoUSA

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