Literature DB >> 22100722

High frequency of fatal haemophagocytic lymphohistiocytosis syndrome in enteropathy-associated T cell lymphoma.

Aurelien Amiot1, Matthieu Allez, Xavier Treton, Claire Fieschi, Lionel Galicier, Francisca Joly, Jean-Marc Gornet, Eric Oksenhendler, Marc Lémann, Yoram Bouhnik.   

Abstract

INTRODUCTION: Enteropathy-associated T-cell lymphoma is a rare form of T-cell lymphoma associated with a poor prognosis and the relative ineffectiveness of standard chemotherapy. The occurrence of haemophagocytic lymphohistiocytosis has been reported only once with this entity. PATIENTS AND METHODS: A retrospective study of 15 patients with enteropathy-associated T-cell lymphoma (type 1 in 12), followed-up in our units, since 1985. Two patients died before starting chemotherapy. The remaining 13 patients were treated with standard chemotherapy (n=7) and purine nucleotide analogues (n=6).
RESULTS: Median follow-up was 8.7 (1-97) months. Surgery was required in 10 patients (66%) for intestinal complications (n=7) or elective small bowel resection (n=3). Survival probability was 40% and 20% at 1 and 5 years, respectively (Kaplan-Meier method). Survival was not significantly different between the two chemotherapy regimens. However, a slight decrease of febrile neutropenia was observed in the purine nucleotide analogues group (p=0.06). Haemophagocytic lymphohistiocytosis occurred in 6/15 (40%) cases. In these six patients, haemophagocytic lymphohistiocytosis was always fatal within 3 months.
CONCLUSION: Enteropathy-associated T-cell lymphoma is associated with a poor outcome, independently of the chemotherapy regimens administered and frequent occurrence of haemophagocytic lymphohistiocytosis. The latter complication should be considered for urgent rescue therapy. Copyright Â
© 2011 Editrice Gastroenterologica Italiana S.r.l. Published by Elsevier Ltd. All rights reserved.

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Year:  2011        PMID: 22100722     DOI: 10.1016/j.dld.2011.10.008

Source DB:  PubMed          Journal:  Dig Liver Dis        ISSN: 1590-8658            Impact factor:   4.088


  5 in total

Review 1.  Lymphoma presenting as secondary HLH: a review with a tale of two cases.

Authors:  Abhimanyu Ghose; Mahender Yellu; Trisha Wise-Draper; Divya Sharma; Julianne Qualtieri; Tahir Latif; Stephen Medlin
Journal:  Clin Lymphoma Myeloma Leuk       Date:  2014-06-21

Review 2.  Updates of Peripheral T Cell Lymphomas Based on the 2017 WHO Classification.

Authors:  Parwiz J Siaghani; Joo Y Song
Journal:  Curr Hematol Malig Rep       Date:  2018-02       Impact factor: 4.213

3.  Hemophagocytic Lymphohistiocytic Syndrome and Enteropathy-Associated T-cell Lymphoma in a Patient with Refractory Celiac Disease.

Authors:  Lucy Lu; Shuoyan Ning; Zain Kassam; Richard Hunt; Marco Puglia
Journal:  ACG Case Rep J       Date:  2014-04-04

4.  A fatal outcome in a patient with coeliac disease who suspended a strict gluten-free diet: a case report.

Authors:  Chong Lu; Xiao-Juan Zheng; Hong-Jun Hua; Qun-Ying Wang
Journal:  J Int Med Res       Date:  2021-12       Impact factor: 1.671

5.  Hemophagocytic lymphohistiocytosis responding to withdrawal of gluten: a case report.

Authors:  Nicholas J Fordham; Richa Ajitsaria; Leena Karnik; Subarna Chakravorty
Journal:  J Med Case Rep       Date:  2016-09-22
  5 in total

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