Literature DB >> 20581392

Everolimus in patients with autosomal dominant polycystic kidney disease.

Gerd Walz1, Klemens Budde, Marwan Mannaa, Jens Nürnberger, Christoph Wanner, Claudia Sommerer, Ulrich Kunzendorf, Bernhard Banas, Walter H Hörl, Nicholas Obermüller, Wolfgang Arns, Hermann Pavenstädt, Jens Gaedeke, Martin Büchert, Christoph May, Harald Gschaidmeier, Stefan Kramer, Kai-Uwe Eckardt.   

Abstract

BACKGROUND: Autosomal dominant polycystic kidney disease (ADPKD) is a slowly progressive hereditary disorder that usually leads to end-stage renal disease. Although the underlying gene mutations were identified several years ago, efficacious therapy to curtail cyst growth and prevent renal failure is not available. Experimental and observational studies suggest that the mammalian target of rapamycin (mTOR) pathway plays a critical role in cyst growth.
METHODS: In this 2-year, double-blind trial, we randomly assigned 433 patients with ADPKD to receive either placebo or the mTOR inhibitor everolimus. The primary outcome was the change in total kidney volume, as measured on magnetic resonance imaging, at 12 and 24 months.
RESULTS: Total kidney volume increased between baseline and 1 year by 102 ml in the everolimus group, versus 157 ml in the placebo group (P=0.02) and between baseline and 2 years by 230 ml and 301 ml, respectively (P=0.06). Cyst volume increased by 76 ml in the everolimus group and 98 ml in the placebo group after 1 year (P=0.27) and by 181 ml and 215 ml, respectively, after 2 years (P=0.28). Parenchymal volume increased by 26 ml in the everolimus group and 62 ml in the placebo group after 1 year (P=0.003) and by 56 ml and 93 ml, respectively, after 2 years (P=0.11). The mean decrement in the estimated glomerular filtration rate after 24 months was 8.9 ml per minute per 1.73 m2 of body-surface area in the everolimus group versus 7.7 ml per minute in the placebo group (P=0.15). Drug-specific adverse events were more common in the everolimus group; the rate of infection was similar in the two groups.
CONCLUSIONS: Within the 2-year study period,as compared with placebo, everolimus slowed the increase in total kidney volume of patients with ADPKD but did not slow the progression of renal impairment [corrected]. (Funded by Novartis; EudraCT number, 2006-001485-16; ClinicalTrials.gov number, NCT00414440.)

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Year:  2010        PMID: 20581392     DOI: 10.1056/NEJMoa1003491

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  235 in total

1.  Scattered Deletion of PKD1 in Kidneys Causes a Cystic Snowball Effect and Recapitulates Polycystic Kidney Disease.

Authors:  Wouter N Leonhard; Malu Zandbergen; Kimberley Veraar; Susan van den Berg; Louise van der Weerd; Martijn Breuning; Emile de Heer; Dorien J M Peters
Journal:  J Am Soc Nephrol       Date:  2014-10-31       Impact factor: 10.121

2.  Angiotensin blockade in late autosomal dominant polycystic kidney disease.

Authors:  Vicente E Torres; Kaleab Z Abebe; Arlene B Chapman; Robert W Schrier; William E Braun; Theodore I Steinman; Franz T Winklhofer; Godela Brosnahan; Peter G Czarnecki; Marie C Hogan; Dana C Miskulin; Frederic F Rahbari-Oskoui; Jared J Grantham; Peter C Harris; Michael F Flessner; Charity G Moore; Ronald D Perrone
Journal:  N Engl J Med       Date:  2014-11-15       Impact factor: 91.245

3.  2-Hydroxyestradiol slows progression of experimental polycystic kidney disease.

Authors:  Sharon Anderson; Terry T Oyama; Jessie N Lindsley; William E Schutzer; Douglas R Beard; Vincent H Gattone; Radko Komers
Journal:  Am J Physiol Renal Physiol       Date:  2011-12-07

4.  Primary cilia regulate mTORC1 activity and cell size through Lkb1.

Authors:  Christopher Boehlke; Fruzsina Kotsis; Vishal Patel; Simone Braeg; Henriette Voelker; Saskia Bredt; Theresa Beyer; Heike Janusch; Christoph Hamann; Markus Gödel; Klaus Müller; Martin Herbst; Miriam Hornung; Mara Doerken; Michael Köttgen; Roland Nitschke; Peter Igarashi; Gerd Walz; E Wolfgang Kuehn
Journal:  Nat Cell Biol       Date:  2010-10-24       Impact factor: 28.824

5.  Do mTOR inhibitors still have a future in ADPKD?

Authors:  Norberto Perico; Giuseppe Remuzzi
Journal:  Nat Rev Nephrol       Date:  2010-12       Impact factor: 28.314

6.  Everolimus-related organizing pneumonia: a report establishing causality.

Authors:  Justine Frija; Dominique Joly; Bertrand Knebelmann; Daniel Dusser; Pierre-Régis Burgel
Journal:  Invest New Drugs       Date:  2010-12-29       Impact factor: 3.850

7.  Rapamycin-mediated suppression of renal cyst expansion in del34 Pkd1-/- mutant mouse embryos: an investigation of the feasibility of renal cyst prevention in the foetus.

Authors:  Cherie Stayner; Justin Shields; Lynn Slobbe; Jonathan M Shillingford; Thomas Weimbs; Michael R Eccles
Journal:  Nephrology (Carlton)       Date:  2012-11       Impact factor: 2.506

8.  Disorders of fatty acid oxidation and autosomal recessive polycystic kidney disease-different clinical entities and comparable perinatal renal abnormalities.

Authors:  Agnes Hackl; Katrin Mehler; Ingo Gottschalk; Anne Vierzig; Marcus Eydam; Jan Hauke; Bodo B Beck; Max C Liebau; Regina Ensenauer; Lutz T Weber; Sandra Habbig
Journal:  Pediatr Nephrol       Date:  2017-01-12       Impact factor: 3.714

Review 9.  Vasopressin-2 receptor signaling and autosomal dominant polycystic kidney disease: from bench to bedside and back again.

Authors:  Markus M Rinschen; Bernhard Schermer; Thomas Benzing
Journal:  J Am Soc Nephrol       Date:  2014-02-20       Impact factor: 10.121

Review 10.  The importance of total kidney volume in evaluating progression of polycystic kidney disease.

Authors:  Jared J Grantham; Vicente E Torres
Journal:  Nat Rev Nephrol       Date:  2016-10-03       Impact factor: 28.314

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