Literature DB >> 25810439

Translating genetic findings in hereditary nephrotic syndrome: the missing loops.

Gentzon Hall1, Rasheed A Gbadegesin2.   

Abstract

Nephrotic syndrome (NS) is a clinicopathological entity characterized by proteinuria, hypoalbuminemia, peripheral edema, and hyperlipidemia. It is the most common cause of glomerular disease in children and adults. Although the molecular pathogenesis of NS is not completely understood, data from the study of familial NS suggest that it is a "podocytopathy." Virtually all of the genes mutated in hereditary NS localize to the podocyte or its secreted products and the slit diaphragm. Since the completion of human genome sequence and the advent of next generation sequencing, at least 29 causes of single-gene NS have been identified. However, these findings have not been matched by therapeutic advances owing to suboptimal in vitro and in vivo models for the study of human glomerular disease and podocyte injury phenotypes. Multidisciplinary collaboration between clinicians, geneticists, cell biologists, and molecular physiologists has the potential to overcome this barrier and thereby speed up the translation of genetic findings into improved patient care.
Copyright © 2015 the American Physiological Society.

Entities:  

Keywords:  gene mutation; nephrotic syndrome; podocyte

Mesh:

Year:  2015        PMID: 25810439      PMCID: PMC4490379          DOI: 10.1152/ajprenal.00683.2014

Source DB:  PubMed          Journal:  Am J Physiol Renal Physiol        ISSN: 1522-1466


  41 in total

Review 1.  Educational paper: the podocytopathies.

Authors:  Anja K Büscher; Stefanie Weber
Journal:  Eur J Pediatr       Date:  2012-01-13       Impact factor: 3.183

2.  NPHS2, encoding the glomerular protein podocin, is mutated in autosomal recessive steroid-resistant nephrotic syndrome.

Authors:  N Boute; O Gribouval; S Roselli; F Benessy; H Lee; A Fuchshuber; K Dahan; M C Gubler; P Niaudet; C Antignac
Journal:  Nat Genet       Date:  2000-04       Impact factor: 38.330

3.  Mutations in the gene that encodes the F-actin binding protein anillin cause FSGS.

Authors:  Rasheed A Gbadegesin; Gentzon Hall; Adebowale Adeyemo; Nils Hanke; Irini Tossidou; James Burchette; Guanghong Wu; Alison Homstad; Matthew A Sparks; Jose Gomez; Ruiji Jiang; Andrea Alonso; Peter Lavin; Peter Conlon; Ron Korstanje; M Christine Stander; Ghaidan Shamsan; Moumita Barua; Robert Spurney; Pravin C Singhal; Jeffrey B Kopp; Hermann Haller; David Howell; Martin R Pollak; Andrey S Shaw; Mario Schiffer; Michelle P Winn
Journal:  J Am Soc Nephrol       Date:  2014-03-27       Impact factor: 10.121

4.  Glucocorticoids protect and enhance recovery of cultured murine podocytes via actin filament stabilization.

Authors:  Richard F Ransom; Nancy G Lam; Mark A Hallett; Simon J Atkinson; William E Smoyer
Journal:  Kidney Int       Date:  2005-12       Impact factor: 10.612

Review 5.  Genetic testing in nephrotic syndrome--challenges and opportunities.

Authors:  Rasheed A Gbadegesin; Michelle P Winn; William E Smoyer
Journal:  Nat Rev Nephrol       Date:  2013-01-15       Impact factor: 28.314

Review 6.  TRPC6 and FSGS: the latest TRP channelopathy.

Authors:  Nirvan Mukerji; Tirupapuliyur V Damodaran; Michelle P Winn
Journal:  Biochim Biophys Acta       Date:  2007-03-20

Review 7.  The zebrafish pronephros: a model to study nephron segmentation.

Authors:  R A Wingert; A J Davidson
Journal:  Kidney Int       Date:  2008-03-05       Impact factor: 10.612

8.  Positionally cloned gene for a novel glomerular protein--nephrin--is mutated in congenital nephrotic syndrome.

Authors:  M Kestilä; U Lenkkeri; M Männikkö; J Lamerdin; P McCready; H Putaala; V Ruotsalainen; T Morita; M Nissinen; R Herva; C E Kashtan; L Peltonen; C Holmberg; A Olsen; K Tryggvason
Journal:  Mol Cell       Date:  1998-03       Impact factor: 17.970

9.  Podocyte-secreted angiopoietin-like-4 mediates proteinuria in glucocorticoid-sensitive nephrotic syndrome.

Authors:  Lionel C Clement; Carmen Avila-Casado; Camille Macé; Elizabeth Soria; Winston W Bakker; Sander Kersten; Sumant S Chugh
Journal:  Nat Med       Date:  2010-12-12       Impact factor: 53.440

10.  The fine structure of the renal glomerulus of the mouse.

Authors:  E YAMADA
Journal:  J Biophys Biochem Cytol       Date:  1955-11-25
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  10 in total

1.  Steroid-resistant nephrotic syndrome caused by co-inheritance of mutations at NPHS1 and ADCK4 genes in two Chinese siblings.

Authors:  Hongwen Zhang; Fang Wang; Xiaoyu Liu; Xuhui Zhong; Yong Yao; Huijie Xiao
Journal:  Intractable Rare Dis Res       Date:  2017-11

2.  Evaluating Mendelian nephrotic syndrome genes for evidence for risk alleles or oligogenicity that explain heritability.

Authors:  Brendan D Crawford; Christopher E Gillies; Catherine C Robertson; Matthias Kretzler; Edgar A Otto; Virginia Vega-Warner; Matthew G Sampson
Journal:  Pediatr Nephrol       Date:  2016-10-20       Impact factor: 3.714

3.  Urinary Anomalies in 22q11.2 Deletion (DiGeorge syndrome): From Copy Number Variations to Single-Gene Determinants of Phenotype.

Authors:  Gentzon Hall; Jonathan C Routh; Rasheed A Gbadegesin
Journal:  Am J Kidney Dis       Date:  2017-04-26       Impact factor: 8.860

Review 4.  Treatment of steroid-resistant nephrotic syndrome in the genomic era.

Authors:  Adam R Bensimhon; Anna E Williams; Rasheed A Gbadegesin
Journal:  Pediatr Nephrol       Date:  2018-10-02       Impact factor: 3.714

Review 5.  Lessons from genetics: is it time to revise the therapeutic approach to children with steroid-resistant nephrotic syndrome?

Authors:  Francesca Becherucci; Benedetta Mazzinghi; Aldesia Provenzano; Luisa Murer; Sabrina Giglio; Paola Romagnani
Journal:  J Nephrol       Date:  2016-05-21       Impact factor: 3.902

6.  The Human FSGS-Causing ANLN R431C Mutation Induces Dysregulated PI3K/AKT/mTOR/Rac1 Signaling in Podocytes.

Authors:  Gentzon Hall; Brandon M Lane; Kamal Khan; Igor Pediaditakis; Jianqiu Xiao; Guanghong Wu; Liming Wang; Maria E Kovalik; Megan Chryst-Stangl; Erica E Davis; Robert F Spurney; Rasheed A Gbadegesin
Journal:  J Am Soc Nephrol       Date:  2018-07-12       Impact factor: 10.121

7.  Knockout of TRPC6 promotes insulin resistance and exacerbates glomerular injury in Akita mice.

Authors:  Liming Wang; Jae-Hyung Chang; Anne F Buckley; Robert F Spurney
Journal:  Kidney Int       Date:  2019-02       Impact factor: 10.612

8.  Genetic Testing for Steroid-Resistant-Nephrotic Syndrome in an Outbred Population.

Authors:  Jennifer D Varner; Megan Chryst-Stangl; Christopher Imokhuede Esezobor; Adaobi Solarin; Guanghong Wu; Brandon Lane; Gentzon Hall; Asiri Abeyagunawardena; Ayo Matory; Tracy E Hunley; Jen Jar Lin; David Howell; Rasheed Gbadegesin
Journal:  Front Pediatr       Date:  2018-10-22       Impact factor: 3.418

Review 9.  Prospects of genetic testing for steroid-resistant nephrotic syndrome in Nigerian children: a narrative review of challenges and opportunities.

Authors:  Emmanuel Ademola Anigilaje; Ayodotun Olutola
Journal:  Int J Nephrol Renovasc Dis       Date:  2019-05-08

Review 10.  Diagnostic and Management Challenges in Congenital Nephrotic Syndrome.

Authors:  Ben Christopher Reynolds; Robert James Alan Oswald
Journal:  Pediatric Health Med Ther       Date:  2019-12-17
  10 in total

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