Literature DB >> 22166847

Early angiotensin-converting enzyme inhibition in Alport syndrome delays renal failure and improves life expectancy.

Oliver Gross1, Christoph Licht, Hans J Anders, Bernd Hoppe, Bodo Beck, Burkhard Tönshoff, Britta Höcker, Simone Wygoda, Jochen H H Ehrich, Lars Pape, Martin Konrad, Wolfgang Rascher, Jörg Dötsch, Dirk E Müller-Wiefel, Peter Hoyer, Bertrand Knebelmann, Yves Pirson, Jean-Pierre Grunfeld, Patrick Niaudet, Pierre Cochat, Laurence Heidet, Said Lebbah, Roser Torra, Tim Friede, Katharina Lange, Gerhard A Müller, Manfred Weber.   

Abstract

Alport syndrome inevitably leads to end-stage renal disease and there are no therapies known to improve outcome. Here we determined whether angiotensin-converting enzyme inhibitors can delay time to dialysis and improve life expectancy in three generations of Alport families. Patients were categorized by renal function at the initiation of therapy and included 33 with hematuria or microalbuminuria, 115 with proteinuria, 26 with impaired renal function, and 109 untreated relatives. Patients were followed for a period whose mean duration exceeded two decades. Untreated relatives started dialysis at a median age of 22 years. Treatment of those with impaired renal function significantly delayed dialysis to a median age of 25, while treatment of those with proteinuria delayed dialysis to a median age of 40. Significantly, no patient with hematuria or microalbuminuria advanced to renal failure so far. Sibling pairs confirmed these results, showing that earlier therapy in younger patients significantly delayed dialysis by 13 years compared to later or no therapy in older siblings. Therapy significantly improved life expectancy beyond the median age of 55 years of the no-treatment cohort. Thus, Alport syndrome is treatable with angiotensin-converting enzyme inhibition to delay renal failure and therapy improves life expectancy in a time-dependent manner. This supports the need for early diagnosis and early nephroprotective therapy in oligosymptomatic patients.

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Year:  2011        PMID: 22166847     DOI: 10.1038/ki.2011.407

Source DB:  PubMed          Journal:  Kidney Int        ISSN: 0085-2538            Impact factor:   10.612


  101 in total

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Journal:  CEN Case Rep       Date:  2020-06-26

2.  Alport syndrome: ACEIs delay RRT and increase lifespan in Alport syndrome.

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Journal:  Nat Rev Nephrol       Date:  2012-01-10       Impact factor: 28.314

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Review 4.  The collagenopathies: review of clinical phenotypes and molecular correlations.

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Journal:  Curr Rheumatol Rep       Date:  2014-01       Impact factor: 4.592

5.  Improving mutation screening in familial hematuric nephropathies through next generation sequencing.

Authors:  Vincent Morinière; Karin Dahan; Pascale Hilbert; Marieline Lison; Said Lebbah; Alexandra Topa; Christine Bole-Feysot; Solenn Pruvost; Patrick Nitschke; Emmanuelle Plaisier; Bertrand Knebelmann; Marie-Alice Macher; Laure-Hélène Noel; Marie-Claire Gubler; Corinne Antignac; Laurence Heidet
Journal:  J Am Soc Nephrol       Date:  2014-05-22       Impact factor: 10.121

Review 6.  Ocular features in Alport syndrome: pathogenesis and clinical significance.

Authors:  Judy Savige; Shivanand Sheth; Anita Leys; Anjali Nicholson; Heather G Mack; Deb Colville
Journal:  Clin J Am Soc Nephrol       Date:  2015-02-03       Impact factor: 8.237

7.  Alport syndrome: a rare cause of uraemia.

Authors:  Soumik Ghosh; Manavdeep Singh; Ratnakar Sahoo; Sachin Rao
Journal:  BMJ Case Rep       Date:  2014-02-13

8.  Alport's syndrome with type 4 renal tubular acidosis.

Authors:  Thomas McDonnell; Chukwuma Chukwu; Christopher Wong
Journal:  BMJ Case Rep       Date:  2017-05-31

Review 9.  Collagen IV diseases: A focus on the glomerular basement membrane in Alport syndrome.

Authors:  Dominic Cosgrove; Shiguang Liu
Journal:  Matrix Biol       Date:  2016-08-27       Impact factor: 11.583

Review 10.  An update on the pathomechanisms and future therapies of Alport syndrome.

Authors:  Damien Noone; Christoph Licht
Journal:  Pediatr Nephrol       Date:  2012-08-18       Impact factor: 3.714

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