Cargando…

Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease

Some experimental studies have suggested a beneficial effect of the mammalian target of rapamycin (mTOR) inhibitor use on hepatic and renal cyst growth in patients with autosomal dominant polycystic kidney disease (ADPKD). However, the results of clinical studies are conflicting and the role of mTOR...

Descripción completa

Detalles Bibliográficos
Autores principales: Friedrich, L., Barbey, F., Pascual, M., Venetz, J.-P.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi Publishing Corporation 2012
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3530859/
https://www.ncbi.nlm.nih.gov/pubmed/23304619
http://dx.doi.org/10.1155/2012/513025
_version_ 1782254072145903616
author Friedrich, L.
Barbey, F.
Pascual, M.
Venetz, J.-P.
author_facet Friedrich, L.
Barbey, F.
Pascual, M.
Venetz, J.-P.
author_sort Friedrich, L.
collection PubMed
description Some experimental studies have suggested a beneficial effect of the mammalian target of rapamycin (mTOR) inhibitor use on hepatic and renal cyst growth in patients with autosomal dominant polycystic kidney disease (ADPKD). However, the results of clinical studies are conflicting and the role of mTOR inhibitors is still uncertain. We report the case of a patient with ADPKD who underwent deceased kidney transplantation because of an end-stage renal disease. The evolution was uneventful with an excellent graft function under cyclosporine (CsA) monotherapy. Some years later, the patient developed a symptomatic hepatomegaly due to growth of cysts. CsA was replaced by sirolimus, an mTOR inhibitor, in order to reduce or control the increase in the cyst and liver volume. Despite the switch, the hepatic volume increased by 25% in two years. Finally sirolimus was stopped because of the lack of effect on hepatic cyst growth and the presence of sirolimus side effects. The interest of our case resides in the followup by MRI imaging during the mTOR inhibitor treatment and 15 months after the restart of the initial immunosuppressive therapy. This observation indicates that mTOR inhibitors did not have significant effect on cyst-associated hepatic growth in our patient, which is consistent with some results of recent large clinical studies.
format Online
Article
Text
id pubmed-3530859
institution National Center for Biotechnology Information
language English
publishDate 2012
publisher Hindawi Publishing Corporation
record_format MEDLINE/PubMed
spelling pubmed-35308592013-01-09 Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease Friedrich, L. Barbey, F. Pascual, M. Venetz, J.-P. Case Rep Transplant Case Report Some experimental studies have suggested a beneficial effect of the mammalian target of rapamycin (mTOR) inhibitor use on hepatic and renal cyst growth in patients with autosomal dominant polycystic kidney disease (ADPKD). However, the results of clinical studies are conflicting and the role of mTOR inhibitors is still uncertain. We report the case of a patient with ADPKD who underwent deceased kidney transplantation because of an end-stage renal disease. The evolution was uneventful with an excellent graft function under cyclosporine (CsA) monotherapy. Some years later, the patient developed a symptomatic hepatomegaly due to growth of cysts. CsA was replaced by sirolimus, an mTOR inhibitor, in order to reduce or control the increase in the cyst and liver volume. Despite the switch, the hepatic volume increased by 25% in two years. Finally sirolimus was stopped because of the lack of effect on hepatic cyst growth and the presence of sirolimus side effects. The interest of our case resides in the followup by MRI imaging during the mTOR inhibitor treatment and 15 months after the restart of the initial immunosuppressive therapy. This observation indicates that mTOR inhibitors did not have significant effect on cyst-associated hepatic growth in our patient, which is consistent with some results of recent large clinical studies. Hindawi Publishing Corporation 2012 2012-12-13 /pmc/articles/PMC3530859/ /pubmed/23304619 http://dx.doi.org/10.1155/2012/513025 Text en Copyright © 2012 L. Friedrich et al. https://creativecommons.org/licenses/by/3.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Friedrich, L.
Barbey, F.
Pascual, M.
Venetz, J.-P.
Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease
title Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease
title_full Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease
title_fullStr Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease
title_full_unstemmed Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease
title_short Absence of mTOR Inhibitor Effect on Hepatic Cyst Growth: A Case Report of a Kidney Transplant Recipient with Autosomal Dominant Polycystic Kidney Disease
title_sort absence of mtor inhibitor effect on hepatic cyst growth: a case report of a kidney transplant recipient with autosomal dominant polycystic kidney disease
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3530859/
https://www.ncbi.nlm.nih.gov/pubmed/23304619
http://dx.doi.org/10.1155/2012/513025
work_keys_str_mv AT friedrichl absenceofmtorinhibitoreffectonhepaticcystgrowthacasereportofakidneytransplantrecipientwithautosomaldominantpolycystickidneydisease
AT barbeyf absenceofmtorinhibitoreffectonhepaticcystgrowthacasereportofakidneytransplantrecipientwithautosomaldominantpolycystickidneydisease
AT pascualm absenceofmtorinhibitoreffectonhepaticcystgrowthacasereportofakidneytransplantrecipientwithautosomaldominantpolycystickidneydisease
AT venetzjp absenceofmtorinhibitoreffectonhepaticcystgrowthacasereportofakidneytransplantrecipientwithautosomaldominantpolycystickidneydisease