Literature DB >> 32564281

Nephropathic cystinosis: an update on genetic conditioning.

Rezan Topaloglu1.   

Abstract

Cystinosis is an autosomal recessive lysosomal storage disorder caused by CTNS gene mutations. The CTNS gene encodes the protein cystinosin, which transports free cystine from lysosomes to cytoplasm. In cases of cystinosin deficiency, free cystine accumulates in lysosomes and forms toxic crystals that lead to tissue and organ damage. Since CTNS gene mutations were first described, many variations have been identified that vary according to geographic region, although the phenotype remains the same. Cystinosis is a hereditary disease that can be treated with the cystine-depleting agent cysteamine. Cysteamine slows organ deterioration, but cannot treat renal Fanconi syndrome or prevent eventual kidney failure; therefore, novel treatment modalities for cystinosis are of great interest to researchers. The present review aims to highlight the geographic differences in cystinosis-specifically in terms of its genetic aspects, clinical features, management, and long-term complications.

Entities:  

Keywords:  Clinical features; Cystinosis; Disparities; Genetics; Geographic regions; Management

Mesh:

Substances:

Year:  2020        PMID: 32564281     DOI: 10.1007/s00467-020-04638-9

Source DB:  PubMed          Journal:  Pediatr Nephrol        ISSN: 0931-041X            Impact factor:   3.714


  7 in total

1.  The aminoglycoside geneticin permits translational readthrough of the CTNS W138X nonsense mutation in fibroblasts from patients with nephropathic cystinosis.

Authors:  Emma J Brasell; LeeLee Chu; Reyhan El Kares; Jung Hwa Seo; Robin Loesch; Diana M Iglesias; Paul Goodyer
Journal:  Pediatr Nephrol       Date:  2018-11-09       Impact factor: 3.714

2.  Improvement in the renal prognosis in nephropathic cystinosis.

Authors:  Karlijn J Van Stralen; Francesco Emma; Kitty J Jager; Enrico Verrina; Franz Schaefer; Guido F Laube; Malcolm A Lewis; Elena N Levtchenko
Journal:  Clin J Am Soc Nephrol       Date:  2011-08-25       Impact factor: 8.237

Review 3.  Potential use of stem cells as a therapy for cystinosis.

Authors:  Celine J Rocca; Stephanie Cherqui
Journal:  Pediatr Nephrol       Date:  2018-05-22       Impact factor: 3.714

4.  The first molecular genetics analysis of individuals suffering from nephropatic cystinosis in the Southwestern Iran.

Authors:  Sepideh Shahkarami; Hamid Galehdari; Ali Ahmadzadeh; Mahnaz Babaahmadi; Mohammad Pedram
Journal:  Nefrologia       Date:  2013-02-15       Impact factor: 2.033

5.  Management of bone disease in cystinosis: Statement from an international conference.

Authors:  Katharina Hohenfellner; Frank Rauch; Gema Ariceta; Atif Awan; Justine Bacchetta; Carsten Bergmann; Susanne Bechtold; Noelle Cassidy; Geroges Deschenes; Ewa Elenberg; William A Gahl; Oliver Greil; Erik Harms; Nadine Herzig; Bernd Hoppe; Christian Koeppl; Malcolm A Lewis; Elena Levtchenko; Galina Nesterova; Fernando Santos; Karl P Schlingmann; Aude Servais; Neveen A Soliman; Guenther Steidle; Clodagh Sweeney; Ulrike Treikauskas; Rezan Topaloglu; Alexey Tsygin; Koenraad Veys; Rodo V Vigier; Jozef Zustin; Dieter Haffner
Journal:  J Inherit Metab Dis       Date:  2019-08-05       Impact factor: 4.982

6.  A 57 kB Genomic Deletion Causing CTNS Loss of Function Contributes to the CTNS Mutational Spectrum in the Middle East.

Authors:  Maryam Najafi; Dor Mohammad Kordi Tamandani; Anoush Azarfar; Zeineb Bakey; Farkhondeh Behjati; Dinu Antony; Isabel Schüle; Simin Sadeghi-Bojd; Ehsan Ghayoor Karimiani; Miriam Schmidts
Journal:  Front Pediatr       Date:  2019-03-21       Impact factor: 3.418

Review 7.  Effects of long-term cysteamine treatment in patients with cystinosis.

Authors:  Gema Ariceta; Vincenzo Giordano; Fernando Santos
Journal:  Pediatr Nephrol       Date:  2017-12-19       Impact factor: 3.714

  7 in total
  4 in total

1.  Clinical and genetic characteristics of Tunisian children with infantile nephropathic cystinosis.

Authors:  Mariem El Younsi; Médiha Trabelsi; Sandra Ben Youssef; Inès Ouertani; Yousra Hammi; Ahlem Achour; Faouzi Maazoul; Maher Kharrat; Tahar Gargah; Ridha M'rad
Journal:  Pediatr Nephrol       Date:  2022-04-20       Impact factor: 3.714

2.  A Bartter syndrome patient presenting with severe growth retardation: Answers.

Authors:  Gökçen Erfidan; Demet Alaygut; Özgür Özdemir Şimşek; Seçil Arslansoyu Çamlar; Fatma Mutlubaş; Belde Kasap Demir
Journal:  Pediatr Nephrol       Date:  2022-01-09       Impact factor: 3.714

Review 3.  Biomarkers in Nephropathic Cystinosis: Current and Future Perspectives.

Authors:  Francesco Emma; Giovanni Montini; Marco Pennesi; Licia Peruzzi; Enrico Verrina; Bianca Maria Goffredo; Fabrizio Canalini; David Cassiman; Silvia Rossi; Elena Levtchenko
Journal:  Cells       Date:  2022-06-04       Impact factor: 7.666

4.  Cysteamine exerts in vitro antiviral activity against the SARS-CoV-2 Delta and Omicron variants.

Authors:  Tonino Alonzi; Alessandra Aiello; Federica Repele; Laura Falasca; Massimo Francalancia; Anna Rosa Garbuglia; Giovanni Delogu; Emanuele Nicastri; Mauro Piacentini; Delia Goletti
Journal:  Cell Death Discov       Date:  2022-06-15
  4 in total

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