Genetic studies of focal segmental glomerulosclerosis: a waste of scientific time?

被引:4
作者
Howie, Alexander J. [1 ]
机构
[1] UCL, Royal Free Hosp, Dept Cellular Pathol, Dept Pathol, London NW3 2QG, England
关键词
Focal segmental glomerulosclerosis; Genetics; Renal pathology; NEPHROTIC SYNDROME; PATHOLOGICAL CLASSIFICATION; MITOCHONDRIAL-DNA; STORAGE-DISEASE; RENAL-DISEASE; MUTATIONS; FSGS; SPECTRUM; VARIANT; REPRODUCIBILITY;
D O I
10.1007/s00467-018-4161-6
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Many genetic causes of focal segmental glomerulosclerosis (FSGS) have been described. A paradox is that the science in the molecular biology, which generally appears of high quality, is not mirrored by a similarly critical analysis of the renal pathology. FSGS has been applied to such a wide range of conditions that it can reasonably be said to have no useful meaning. Attempts to refine the term have been largely ignored. Study of 252 papers on genetic causes of FSGS found various clinical features. Many papers took the reported diagnosis without question. Few papers reported a pathological review, almost half reported FSGS and up to six other conditions caused by any particular gene, some reported FSGS with recognisable glomerular disorders, over 80% did not apply the Columbia classification, and in nearly all with photomicrographs, the images were not useful for refinement of FSGS. Some workers commented on a lack of genotype-phenotype correlation. One reason is a disregard of the principle that scientific investigation requires an unambiguous definition of the condition studied, to allow others to replicate or refute the findings. Genetic studies of FSGS should use a similarly rigorous approach to renal pathology to that used in molecular biology.
引用
收藏
页码:9 / 16
页数:8
相关论文
共 66 条
  • [1] Genetic causes of proteinuria and nephrotic syndrome: Impact on podocyte pathobiology
    Akchurin, Oleh
    Reidy, Kimberly J.
    [J]. PEDIATRIC NEPHROLOGY, 2015, 30 (02) : 221 - 233
  • [2] [Anonymous], 2014, PLOS ONE, V9
  • [3] Simultaneous sequencing of 37 genes identified causative mutations in the majority of children with renal tubulopathies
    Ashton, Emma J.
    Legrand, Anne
    Benoit, Valerie
    Roncelin, Isabelle
    Venisse, Annabelle
    Zennaro, Maria-Christina
    Jeunemaitre, Xavier
    Iancu, Daniela
    van't Hoff, William G.
    Walsh, Stephen B.
    Godefroid, Nathalie
    Rotthier, Annelies
    Del Favero, Jurgen
    Devuyst, Olivier
    Schaefer, Franz
    Jenkins, Lucy A.
    Kleta, Robert
    Dahan, Karin
    Vargas-Poussou, Rosa
    Bockenhauer, Detlef
    [J]. KIDNEY INTERNATIONAL, 2018, 93 (04) : 961 - 967
  • [4] HYPERFILTRATION AND RENAL-DISEASE IN GLYCOGEN-STORAGE DISEASE, TYPE-I
    BAKER, L
    DAHLEM, S
    GOLDFARB, S
    KERN, EFO
    STANLEY, CA
    EGLER, J
    OLSHAN, JS
    HEYMAN, S
    [J]. KIDNEY INTERNATIONAL, 1989, 35 (06) : 1345 - 1350
  • [5] Influence of aldosterone synthase gene C-344T polymorphism on focal segmental glomerulosclerosis
    Bantis, Christos
    Heering, Peter J.
    Stangou, Maria
    Kouri, Nicoletta-Maria
    Schwandt, Christina
    Memmos, Dimitrios
    Rump, Lars-Christian
    Ivens, Katrin
    [J]. NEPHROLOGY, 2011, 16 (08) : 730 - 735
  • [6] Reproducibility of the NEPTUNE descriptor-based scoring system on whole-slide images and histologic and ultrastructural digital images
    Barisoni, Laura
    Troost, Jonathan P.
    Nast, Cynthia
    Bagnasco, Serena
    Avila-Casado, Carmen
    Hodgin, Jeffrey
    Palmer, Matthew
    Rosenberg, Avi
    Gasim, Adil
    Liensziewski, Chrysta
    Merlino, Lino
    Chien, Hui-Ping
    Chang, Anthony
    Meehan, Shane M.
    Gaut, Joseph
    Song, Peter
    Holzman, Lawrence
    Gibson, Debbie
    Kretzler, Matthias
    Gillespie, Brenda W.
    Hewitt, Stephen M.
    [J]. MODERN PATHOLOGY, 2016, 29 (07) : 671 - 684
  • [7] Hereditary kidney diseases: highlighting the importance of classical Mendelian phenotypes
    Benoit, Genevieve
    Machuca, Eduardo
    Heidet, Laurence
    Antignac, Corinne
    [J]. YEAR IN HUMAN AND MEDICAL GENETICS: NEW TRENDS IN MENDELIAN GENETICS, 2010, 1214 : 83 - 98
  • [8] Recurrence of focal segmental glomerulosclerosis after renal transplantation in patients with mutations of podocin
    Bertelli, R
    Ginevri, F
    Caridi, G
    Dagnino, M
    Sandrini, S
    Di Duca, M
    Emma, F
    Sanna-Cherchi, S
    Scolari, F
    Neri, TM
    Murer, L
    Massella, L
    Basile, G
    Rizzoni, G
    Perfumo, F
    Ghiggeri, GM
    [J]. AMERICAN JOURNAL OF KIDNEY DISEASES, 2003, 41 (06) : 1314 - 1321
  • [9] Deriving and understanding the risk of post-transplant recurrence of nephrotic syndrome in the light of current molecular and genetic advances
    Bierzynska, Agnieszka
    Saleem, Moin A.
    [J]. PEDIATRIC NEPHROLOGY, 2018, 33 (11) : 2027 - 2035
  • [10] MAGI2 Mutations Cause Congenital Nephrotic Syndrome
    Bierzynska, Agnieszka
    Soderquest, Katrina
    Dean, Philip
    Colby, Elizabeth
    Rollason, Ruth
    Jones, Caroline
    Inward, Carol D.
    McCarthy, Hugh J.
    Simpson, Michael A.
    Lord, Graham M.
    Williams, Maggie
    Welsh, Gavin I.
    Koziell, Ania B.
    Saleem, Moin A.
    [J]. JOURNAL OF THE AMERICAN SOCIETY OF NEPHROLOGY, 2017, 28 (05): : 1614 - 1621