Literature DB >> 21786142

Genetic basis of cystinosis in Turkish patients: a single-center experience.

Rezan Topaloglu1, Thierry Vilboux, Turgay Coskun, Fatih Ozaltin, Brad Tinloy, Meral Gunay-Aygun, Aysin Bakkaloglu, Nesrin Besbas, Lambert van den Heuvel, Robert Kleta, William A Gahl.   

Abstract

We report the molecular findings for the CTNS gene in 12 Turkish cystinosis patients aged 7-29 years. All presented initially with severe failure to thrive, polyuria, and polydipsia. Cystinosis was diagnosed at age 1 month to 9 years. Seven patients reached end-stage renal failure at ages ranging from 6.5 to 15 years. Whereas three of the remaining five have renal Fanconi syndrome with proteinuria, two have had kidney failure of varying degrees. Molecular analyses involved an initial multiplex polymerase chain reaction (PCR) to determine the presence or absence of the 57-kb northern European founder deletion in CTNS, followed by sequencing of the ten coding exons of CTNS. Comprehensive mutation analysis verified that none of the 12 patients carried the common 57-kb deletion. We identified four previously reported nucleotide variations associated with cystinosis and five new variants: a 10-kb deletion, three missense variants, and a nucleotide substitution in a potential branch point site of intron 4. This study is the first molecular analysis of Turkish cystinosis patients and provides guidance for the molecular diagnosis of cystinosis in this population.

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Year:  2011        PMID: 21786142      PMCID: PMC3501933          DOI: 10.1007/s00467-011-1942-6

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


  26 in total

Review 1.  Splicing in action: assessing disease causing sequence changes.

Authors:  D Baralle; M Baralle
Journal:  J Med Genet       Date:  2005-10       Impact factor: 6.318

2.  Increased free-cystine content of fibroblasts cultured from patients with cystinosis.

Authors:  J A Schneider; F M Rosenbloom; K H Bradley; J E Seegmiller
Journal:  Biochem Biophys Res Commun       Date:  1967-11-30       Impact factor: 3.575

3.  Severity of phenotype in cystinosis varies with mutations in the CTNS gene: predicted effect on the model of cystinosin.

Authors:  M Attard; G Jean; L Forestier; S Cherqui; W van't Hoff; M Broyer; C Antignac; M Town
Journal:  Hum Mol Genet       Date:  1999-12       Impact factor: 6.150

4.  Molecular analysis of cystinosis: probable Irish origin of the most common French Canadian mutation.

Authors:  J McGowan-Jordan; K Stoddard; L Podolsky; E Orrbine; P McLaine; M Town; P Goodyer; A MacKenzie; H Heick
Journal:  Eur J Hum Genet       Date:  1999-09       Impact factor: 4.246

5.  Swallowing dysfunction in 101 patients with nephropathic cystinosis: benefit of long-term cysteamine therapy.

Authors:  Barbara C Sonies; Phaedra Almajid; Robert Kleta; Isa Bernardini; William A Gahl
Journal:  Medicine (Baltimore)       Date:  2005-05       Impact factor: 1.889

6.  Cysteamine therapy for children with nephropathic cystinosis.

Authors:  W A Gahl; G F Reed; J G Thoene; J D Schulman; W B Rizzo; A J Jonas; D W Denman; J J Schlesselman; B J Corden; J A Schneider
Journal:  N Engl J Med       Date:  1987-04-16       Impact factor: 91.245

7.  Mutation spectrum of homogentisic acid oxidase (HGD) in alkaptonuria.

Authors:  Thierry Vilboux; Michael Kayser; Wendy Introne; Pim Suwannarat; Isa Bernardini; Roxanne Fischer; Kevin O'Brien; Robert Kleta; Marjan Huizing; William A Gahl
Journal:  Hum Mutat       Date:  2009-12       Impact factor: 4.878

8.  Characterization of CTNS mutations in Arab patients with cystinosis.

Authors:  Mohammed A Aldahmesh; Amal Humeidan; Hamad A Almojalli; Arif O Khan; Mohammed Rajab; Abbas A AL-Abbad; Brian F Meyer; Fowzan S Alkuraya
Journal:  Ophthalmic Genet       Date:  2009-12       Impact factor: 1.803

9.  Nephropathic cystinosis in adults: natural history and effects of oral cysteamine therapy.

Authors:  William A Gahl; Joan Z Balog; Robert Kleta
Journal:  Ann Intern Med       Date:  2007-08-21       Impact factor: 25.391

10.  Analysis of the CTNS gene in nephropathic cystinosis Mexican patients: report of four novel mutations and identification of a false positive 57-kb deletion genotype with LDM-2/exon 4 multiplex PCR assay.

Authors:  Miguel Angel Alcántara-Ortigoza; Leticia Belmont-Martínez; Marcela Vela-Amieva; Ariadna González-Del Angel
Journal:  Genet Test       Date:  2008-09
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  18 in total

1.  Infantile Nephropathic Cystinosis: A Novel CTNS Mutation.

Authors:  Hakan Doneray; Mohammed Aldahmesh; Gulsah Yilmaz; Emine Cinici; Zerrin Orbak
Journal:  Eurasian J Med       Date:  2017-06

2.  Clinical utility gene card for: cystinosis.

Authors:  Elena Levtchenko; Lambertus van den Heuvel; Francesco Emma; Corinne Antignac
Journal:  Eur J Hum Genet       Date:  2013-09-18       Impact factor: 4.246

3.  Next generation sequencing as second-tier test in high-throughput newborn screening for nephropathic cystinosis.

Authors:  Tobias Fleige; Siegfried Burggraf; Ludwig Czibere; Julia Häring; Birgit Glück; Lisa Marie Keitel; Olfert Landt; Erik Harms; Katharina Hohenfellner; Jürgen Durner; Wulf Röschinger; Marc Becker
Journal:  Eur J Hum Genet       Date:  2019-09-30       Impact factor: 4.246

4.  Slow progression of renal failure in a child with infantile cystinosis.

Authors:  Maria Bitsori; Eleni Vergadi; Emmanouil Galanakis
Journal:  CEN Case Rep       Date:  2018-02-14

5.  The Clinical and Mutational Spectrum of Turkish Patients with Cystinosis.

Authors:  Rezan Topaloglu; Bora Gulhan; Mihriban İnözü; Nur Canpolat; Alev Yilmaz; Aytül Noyan; İsmail Dursun; İbrahim Gökçe; Metin Kaya Gürgöze; Nurver Akinci; Esra Baskin; Erkin Serdaroğlu; Beltinge Demircioğlu Kiliç; Selçuk Yüksel; Duygu Övünç Hacihamdioğlu; Emine Korkmaz; Mutlu Hayran; Fatih Ozaltin
Journal:  Clin J Am Soc Nephrol       Date:  2017-08-09       Impact factor: 8.237

6.  Common mutation causes cystinosis in the majority of black South African patients.

Authors:  E Patricia Owen; Jenisha Nandhlal; Felicity Leisegang; George Van der Watt; Peter Nourse; Priya Gajjar
Journal:  Pediatr Nephrol       Date:  2014-10-18       Impact factor: 3.714

7.  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

8.  Mutational Spectrum of the CTNS Gene in Egyptian Patients with Nephropathic Cystinosis.

Authors:  Neveen A Soliman; Mohamed A Elmonem; Lambertus van den Heuvel; Rehab H Abdel Hamid; Mohamed Gamal; Inge Bongaers; Sandrine Marie; Elena Levtchenko
Journal:  JIMD Rep       Date:  2014-01-25

9.  More than tubular dysfunction: cystinosis and kidney outcomes.

Authors:  Bahriye Atmis; Aysun K Bayazit; Derya Cevizli; Deniz Kor; Hatice Busra Fidan; Atil Bisgin; Sebile Kilavuz; Ilker Unal; Kivilcim Eren Erdogan; Engin Melek; Gulfiliz Gonlusen; Ali Anarat; Neslihan Onenli Mungan
Journal:  J Nephrol       Date:  2021-06-07       Impact factor: 3.902

10.  Genetic basis of cystinosis in Tunisian patients: Identification of novel mutation in CTNS gene.

Authors:  Latifa Chkioua; Souhir Khedhiri; Oussama Grissa; Chaker Aloui; Hadhami Ben Turkia; Salima Ferchichi; Abdelhedi Miled; Roseline Froissart; Cecile Acquaviva; Sandrine Laradi
Journal:  Meta Gene       Date:  2015-07-25
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