Literature DB >> 32533415

A rare case of nephrotic syndrome associated with Dent's disease: a case report.

Shinichi Makino1, Maulana A Empitu1,2, Toshimasa Naito1, Masayoshi Ishii1, Hanae Wakabayashi1, Chiwei Lee1, Masashi Aizawa1, Katsuhiko Asanuma3.   

Abstract

Dent's disease is a rare X-linked condition caused by a mutation in CLCN5 and OCRL gene, which impair the megalin-cubilin receptor-mediated endocytosis in kidney's proximal tubules. Thus, it may manifest as nephrotic-range low-molecular-weight proteinuria (LMWP). On the other hand, glomerular proteinuria, hypoalbuminemia, and edema formation are the key features of nephrotic syndrome that rarely found in Dent's disease. Here, we reported a man in his 30 s with Dent's disease presented with leg edema for 5 days. The laboratory results revealed hypoalbuminemia and a decrease of urine β2-microglobulin/urine protein ratio (Uβ2-MG /UP), indicating that the primary origin of proteinuria shifted from LMWP to glomerular proteins. The kidney biopsy revealed glomerular abnormality and calcium deposition in the renal medulla. Electron microscopy of the kidney tissue indicated extensive foot-process effacement of the glomerular podocytes and degeneration of tubular epithelium. After a combination of treatment with prednisolone and cyclosporine (CyA), the nephrotic syndrome was remitted. Given the atypical clinical presentation and the shift of LMWP to glomerular proteinuria in this patient, glomerulopathy and the Dent's disease existed separately in this patient.

Entities:  

Keywords:  Dent's disease; Low-molecular-weight proteinuria (LMWP); Nephrotic syndrome; β2-microglobulin

Year:  2020        PMID: 32533415      PMCID: PMC7502094          DOI: 10.1007/s13730-020-00491-9

Source DB:  PubMed          Journal:  CEN Case Rep        ISSN: 2192-4449


  13 in total

1.  Tubular proteinuria defined by a study of Dent's (CLCN5 mutation) and other tubular diseases.

Authors:  A G Norden; S J Scheinman; M M Deschodt-Lanckman; M Lapsley; J L Nortier; R V Thakker; R J Unwin; O Wrong
Journal:  Kidney Int       Date:  2000-01       Impact factor: 10.612

2.  Prevalence of low molecular weight proteinuria and Dent disease 1 CLCN5 mutations in proteinuric cohorts.

Authors:  Lada Beara-Lasic; Andrea Cogal; Kristin Mara; Felicity Enders; Ramila A Mehta; Zejfa Haskic; Susan L Furth; Howard Trachtman; Steven J Scheinman; Dawn S Milliner; David S Goldfarb; Peter C Harris; John C Lieske
Journal:  Pediatr Nephrol       Date:  2019-03-10       Impact factor: 3.714

3.  A patient with nephrotic-range proteinuria and focal global glomerulosclerosis.

Authors:  Fernando C Fervenza
Journal:  Clin J Am Soc Nephrol       Date:  2013-07-25       Impact factor: 8.237

4.  Glomerular Pathology in Dent Disease and Its Association with Kidney Function.

Authors:  Xiangling Wang; Franca Anglani; Lada Beara-Lasic; Anila J Mehta; Lisa E Vaughan; Loren Herrera Hernandez; Andrea Cogal; Steven J Scheinman; Gema Ariceta; Robert Isom; Lawrence Copelovitch; Felicity T Enders; Dorella Del Prete; Giuseppe Vezzoli; Fabio Paglialonga; Peter C Harris; John C Lieske
Journal:  Clin J Am Soc Nephrol       Date:  2016-10-03       Impact factor: 8.237

Review 5.  Dent's disease.

Authors:  Olivier Devuyst; Rajesh V Thakker
Journal:  Orphanet J Rare Dis       Date:  2010-10-14       Impact factor: 4.123

6.  Hypothesis: Dent disease is an underrecognized cause of focal glomerulosclerosis.

Authors:  Lawrence Copelovitch; Martin A Nash; Bernard S Kaplan
Journal:  Clin J Am Soc Nephrol       Date:  2007-08-08       Impact factor: 8.237

7.  Comparison of retinol-binding protein and beta 2-microglobulin determination in urine for the early detection of tubular proteinuria.

Authors:  A M Bernard; D Moreau; R Lauwerys
Journal:  Clin Chim Acta       Date:  1982-11-24       Impact factor: 3.786

8.  Differentiation of glomerular, tubular, and normal proteinuria: determinations of urinary excretion of beta-2-macroglobulin, albumin, and total protein.

Authors:  P A Peterson; P E Evrin; I Berggård
Journal:  J Clin Invest       Date:  1969-07       Impact factor: 14.808

9.  Involvement of the tubular ClC-type exchanger ClC-5 in glomeruli of human proteinuric nephropathies.

Authors:  Monica Ceol; Emilia Tiralongo; Hans J Baelde; Daniela Vianello; Giovanni Betto; Annunziata Marangelli; Luciana Bonfante; Marialuisa Valente; Mila Della Barbera; Angela D'Angelo; Franca Anglani; Dorella Del Prete
Journal:  PLoS One       Date:  2012-09-24       Impact factor: 3.240

Review 10.  Proteinuria in Dent disease: a review of the literature.

Authors:  Youri van Berkel; Michael Ludwig; Joanna A E van Wijk; Arend Bökenkamp
Journal:  Pediatr Nephrol       Date:  2016-10-18       Impact factor: 3.714

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