Literature DB >> 35098431

A case of Dent disease type 2 with large deletion of OCRL diagnosed after close examination of a school urinary test.

Yaeko Motoyoshi1, Tomoo Yabuuchi2, Kenichiro Miura2, Motoshi Hattori2, Koji Kiyohara3.   

Abstract

A 7-year-old boy visited our hospital for a detailed examination of proteinuria identified in a school urinary test. He had short stature, misaligned teeth, and mild intellectual disability. A urinary examination identified mild proteinuria and extremely high levels of beta-2 microglobulin. On blood examination, his protein, albumin, and creatinine levels were found to be normal; however, his lactate dehydrogenase and creatinine phosphokinase levels were slightly elevated. Upon histological examination, no abnormalities in glomeruli or tubules were found. Considering these results, we diagnosed our patient with Dent disease type 2 (DD2). Although the whole exome sequencing revealed large deletion of OCRL, which was seen only in Lowe syndrome and not in DD2 previously, our final diagnosis for the patient is DD2. A phenotypic continuum exists between Dent disease and Lowe syndrome, and several factors modify the phenotypes caused by defects in OCRL. Although patients have thus far been diagnosed with DD2 or Lowe syndrome on the basis of their symptoms, accumulation and analysis of cases with OCRL defects may hereafter enable more accurate diagnoses.
© 2022. The Author(s) under exclusive licence to The Japan Society of Nephrology.

Entities:  

Keywords:  Congenital cataract; Dent disease type 2; Low molecular weight proteinuria; Lowe syndrome; OCRL

Mesh:

Substances:

Year:  2022        PMID: 35098431      PMCID: PMC9343535          DOI: 10.1007/s13730-022-00685-3

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


  11 in total

1.  From Lowe syndrome to Dent disease: correlations between mutations of the OCRL1 gene and clinical and biochemical phenotypes.

Authors:  Haifa Hichri; John Rendu; Nicole Monnier; Charles Coutton; Olivier Dorseuil; Rosa Vargas Poussou; Geneviève Baujat; Anne Blanchard; François Nobili; Bruno Ranchin; Michel Remesy; Rémi Salomon; Véronique Satre; Joel Lunardi
Journal:  Hum Mutat       Date:  2011-03-10       Impact factor: 4.878

2.  Novel OCRL1 mutations in patients with the phenotype of Dent disease.

Authors:  Boris Utsch; Arend Bökenkamp; Marcus R Benz; Nesrin Besbas; Jörg Dötsch; Ingo Franke; Stefan Fründ; Faysal Gok; Bernd Hoppe; Stephanie Karle; Eberhard Kuwertz-Bröking; Guido Laube; Margarita Neb; Matti Nuutinen; Fatih Ozaltin; Wolfgang Rascher; Troels Ring; Velibor Tasic; Joanna A E van Wijk; Michael Ludwig
Journal:  Am J Kidney Dis       Date:  2006-12       Impact factor: 8.860

Review 3.  Lowe syndrome/Dent-2 disease: A comprehensive review of known and novel aspects.

Authors:  Florian Recker; Heiko Reutter; Michael Ludwig
Journal:  J Pediatr Genet       Date:  2013-06

4.  OCRL1 mutations in Dent 2 patients suggest a mechanism for phenotypic variability.

Authors:  Antony E Shrimpton; Richard R Hoopes; Stephen J Knohl; Paul Hueber; Anita A C Reed; Paul T Christie; Takashi Igarashi; Philip Lee; Anna Lehman; Colin White; David V Milford; Manuel Rivero Sanchez; Robert Unwin; Oliver M Wrong; Rajesh V Thakker; Steven J Scheinman
Journal:  Nephron Physiol       Date:  2009-04-18

5.  OCRL-mutated fibroblasts from patients with Dent-2 disease exhibit INPP5B-independent phenotypic variability relatively to Lowe syndrome cells.

Authors:  Rodrick Montjean; Rifdat Aoidi; Pierrette Desbois; Julien Rucci; Michaël Trichet; Rémi Salomon; John Rendu; Julien Fauré; Joël Lunardi; Gérard Gacon; Pierre Billuart; Olivier Dorseuil
Journal:  Hum Mol Genet       Date:  2014-10-09       Impact factor: 6.150

6.  Dent-2 disease: a mild variant of Lowe syndrome.

Authors:  Arend Bökenkamp; Detlef Böckenhauer; Hae Il Cheong; Bernd Hoppe; Velibor Tasic; Robert Unwin; Michael Ludwig
Journal:  J Pediatr       Date:  2009-07       Impact factor: 4.406

Review 7.  The oculocerebrorenal syndrome of Lowe: an update.

Authors:  Arend Bökenkamp; Michael Ludwig
Journal:  Pediatr Nephrol       Date:  2016-03-24       Impact factor: 3.714

8.  Splicing Analysis of Exonic OCRL Mutations Causing Lowe Syndrome or Dent-2 Disease.

Authors:  Lorena Suarez-Artiles; Ana Perdomo-Ramirez; Elena Ramos-Trujillo; Felix Claverie-Martin
Journal:  Genes (Basel)       Date:  2018-01-04       Impact factor: 4.096

9.  All known patient mutations in the ASH-RhoGAP domains of OCRL affect targeting and APPL1 binding.

Authors:  Heather J McCrea; Summer Paradise; Livia Tomasini; Maria Addis; Maria Antonietta Melis; Maria Antonietta De Matteis; Pietro De Camilli
Journal:  Biochem Biophys Res Commun       Date:  2008-02-26       Impact factor: 3.575

Review 10.  Genetics and phenotypic heterogeneity of Dent disease: the dark side of the moon.

Authors:  Lisa Gianesello; Dorella Del Prete; Franca Anglani; Lorenzo A Calò
Journal:  Hum Genet       Date:  2020-08-29       Impact factor: 4.132

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