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Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report

BACKGROUND: Post-transplant lymphoproliferative disease is a recognized complication following solid organ transplantation. This is usually a B cell disease and frequently associated with Epstein Barr virus infection, although T cell PTLD can occur. T cell PTLD is usually a monomorphic, lymphomatous...

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Autores principales: Glover, Emily K., Speight, R. Alexander, Televantou, Despina, Needham, Stephanie, Sheerin, Neil S.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7285536/
https://www.ncbi.nlm.nih.gov/pubmed/32522162
http://dx.doi.org/10.1186/s12882-020-01884-9
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author Glover, Emily K.
Speight, R. Alexander
Televantou, Despina
Needham, Stephanie
Sheerin, Neil S.
author_facet Glover, Emily K.
Speight, R. Alexander
Televantou, Despina
Needham, Stephanie
Sheerin, Neil S.
author_sort Glover, Emily K.
collection PubMed
description BACKGROUND: Post-transplant lymphoproliferative disease is a recognized complication following solid organ transplantation. This is usually a B cell disease and frequently associated with Epstein Barr virus infection, although T cell PTLD can occur. T cell PTLD is usually a monomorphic, lymphomatous disease associated with an adverse prognosis. CASE REPORT: We report a 52 year old male pre-emptive renal transplant recipient who developed severe diarrhea with weight loss following intensification of his immunosuppression due to antibody mediated rejection 3 years after transplantation. Duodenal biopsy demonstrated monoclonal CD8+ T cell duodenitis leading to increased intraepithlieal lymphocytes and sub-total villous atrophy mimicking coeliac disease. Coeliac disease was excluded by negative anti-tissue transglutaminase antibody, HLA-DQ2 and HLA-DQ8 testing. There was no evidence of lymphoma either on biopsy or CT enterography and no FDG avid disease on PET. Symptoms did not improve with reduction of immunosuppression, but resolved fully on complete withdrawal of treatment. The transplant failed and he was established on dialysis. The diagnosis was early PTLD. CONCLUSIONS: Oesophagogastroduodenoscopy with small bowel biopsies is a useful investigation for determining the cause of diarrhoea in renal transplant patients when more common causes have been excluded. This is the first report that we are aware of clonal T cell PTLD mimicking coeliac disease which only resolved after complete withdrawal of immunosuppression. As treatments for lymphoma are aggressive they are only initiated in the malignant phase and management of early stage PTLD is to minimise risk of progression by reducing immunosuppression. Any plans to retransplant will have to take into consideration the possibility that PTLD will recur.
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spelling pubmed-72855362020-06-10 Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report Glover, Emily K. Speight, R. Alexander Televantou, Despina Needham, Stephanie Sheerin, Neil S. BMC Nephrol Case Report BACKGROUND: Post-transplant lymphoproliferative disease is a recognized complication following solid organ transplantation. This is usually a B cell disease and frequently associated with Epstein Barr virus infection, although T cell PTLD can occur. T cell PTLD is usually a monomorphic, lymphomatous disease associated with an adverse prognosis. CASE REPORT: We report a 52 year old male pre-emptive renal transplant recipient who developed severe diarrhea with weight loss following intensification of his immunosuppression due to antibody mediated rejection 3 years after transplantation. Duodenal biopsy demonstrated monoclonal CD8+ T cell duodenitis leading to increased intraepithlieal lymphocytes and sub-total villous atrophy mimicking coeliac disease. Coeliac disease was excluded by negative anti-tissue transglutaminase antibody, HLA-DQ2 and HLA-DQ8 testing. There was no evidence of lymphoma either on biopsy or CT enterography and no FDG avid disease on PET. Symptoms did not improve with reduction of immunosuppression, but resolved fully on complete withdrawal of treatment. The transplant failed and he was established on dialysis. The diagnosis was early PTLD. CONCLUSIONS: Oesophagogastroduodenoscopy with small bowel biopsies is a useful investigation for determining the cause of diarrhoea in renal transplant patients when more common causes have been excluded. This is the first report that we are aware of clonal T cell PTLD mimicking coeliac disease which only resolved after complete withdrawal of immunosuppression. As treatments for lymphoma are aggressive they are only initiated in the malignant phase and management of early stage PTLD is to minimise risk of progression by reducing immunosuppression. Any plans to retransplant will have to take into consideration the possibility that PTLD will recur. BioMed Central 2020-06-10 /pmc/articles/PMC7285536/ /pubmed/32522162 http://dx.doi.org/10.1186/s12882-020-01884-9 Text en © The Author(s) 2020 Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Glover, Emily K.
Speight, R. Alexander
Televantou, Despina
Needham, Stephanie
Sheerin, Neil S.
Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report
title Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report
title_full Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report
title_fullStr Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report
title_full_unstemmed Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report
title_short Immunosuppression-induced clonal T-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report
title_sort immunosuppression-induced clonal t-cell lymphoproliferative disease causing severe diarrhoea mimicking coeliac disease following renal transplantation: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7285536/
https://www.ncbi.nlm.nih.gov/pubmed/32522162
http://dx.doi.org/10.1186/s12882-020-01884-9
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