Literature DB >> 25297642

Novel mutation of OCRL1 in Lowe syndrome.

Ting Liu1, Zhihui Yue, Haiyan Wang, Huajuan Tong, Liangzhong Sun.   

Abstract

Lowe syndrome is a rare, X-linked recessive genetic disease with multi-organ involvement. The pathogenic gene is OCRL1. The authors analyzed the OCRL1 mutation and summarized the clinical features of a Chinese child with Lowe syndrome. The patient is a 3 year 7 mo-old boy. He presented with hypotonia at birth and gradually presented with bilateral congenital cataracts, psychomotor retardation, hypophosphatemic rickets and renal tubular function disorder. Sequence analysis of OCRL1 revealed a novel insertion mutation, c.2367insA (p. Ala813X), in exon 22. This mutation was suspected to cause a premature stop codon of OCRL1 and truncation of the OCRL1 protein. His mother, who carried a heterozygous mutation, had no sign of abnormality.

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Year:  2014        PMID: 25297642     DOI: 10.1007/s12098-014-1581-6

Source DB:  PubMed          Journal:  Indian J Pediatr        ISSN: 0019-5456            Impact factor:   1.967


  9 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

Review 2.  Inositol 5-phosphatases: insights from the Lowe syndrome protein OCRL.

Authors:  Michelle Pirruccello; Pietro De Camilli
Journal:  Trends Biochem Sci       Date:  2012-02-28       Impact factor: 13.807

3.  Spectrum of mutations in the OCRL1 gene in the Lowe oculocerebrorenal syndrome.

Authors:  T Lin; B M Orrison; A M Leahey; S F Suchy; D J Bernard; R A Lewis; R L Nussbaum
Journal:  Am J Hum Genet       Date:  1997-06       Impact factor: 11.025

4.  Clinical pictures and novel mutations of WT1-associated Denys-Drash syndrome in two Chinese children.

Authors:  Zhihui Yue; Yuanyuan Pei; Liangzhong Sun; Weijun Huang; Han Huang; Bin Hu; Juan Yang; Xiaoyun Jiang; Ying Mo; Shumei Chen; Kar Neng Lai; Yiming Wang
Journal:  Ren Fail       Date:  2011-08-18       Impact factor: 2.606

5.  Carrier assessment in families with lowe oculocerebrorenal syndrome: novel mutations in the OCRL1 gene and correlation of direct DNA diagnosis with ocular examination.

Authors:  W Röschinger; A C Muntau; G Rudolph; A A Roscher; S Kammerer
Journal:  Mol Genet Metab       Date:  2000-03       Impact factor: 4.797

6.  Renal manifestations of Dent disease and Lowe syndrome.

Authors:  Hee Yeon Cho; Bum Hee Lee; Hyun Jin Choi; Il Soo Ha; Yong Choi; Hae Il Cheong
Journal:  Pediatr Nephrol       Date:  2007-11-24       Impact factor: 3.714

7.  Renal phenotype in Lowe Syndrome: a selective proximal tubular dysfunction.

Authors:  Detlef Bockenhauer; Arend Bokenkamp; William van't Hoff; Elena Levtchenko; Joana E Kist-van Holthe; Velibor Tasic; Michael Ludwig
Journal:  Clin J Am Soc Nephrol       Date:  2008-05-14       Impact factor: 8.237

8.  Molecular confirmation of carriers for Lowe syndrome.

Authors:  T Lin; R A Lewis; R L Nussbaum
Journal:  Ophthalmology       Date:  1999-01       Impact factor: 12.079

Review 9.  Lowe syndrome.

Authors:  Mario Loi
Journal:  Orphanet J Rare Dis       Date:  2006-05-18       Impact factor: 4.123

  9 in total
  3 in total

1.  Lowe syndrome patient cells display mTOR- and RhoGTPase-dependent phenotypes alleviated by rapamycin and statins.

Authors:  Kayalvizhi Madhivanan; Swetha Ramadesikan; Wen-Chieh Hsieh; Mariana C Aguilar; Claudia B Hanna; Robert L Bacallao; R Claudio Aguilar
Journal:  Hum Mol Genet       Date:  2020-06-27       Impact factor: 6.150

2.  Ocular Pathology of Oculocerebrorenal Syndrome of Lowe: Novel Mutations and Genotype-Phenotype Analysis.

Authors:  Emilie Song; Na Luo; Jorge A Alvarado; Maria Lim; Cathleen Walnuss; Daniel Neely; Dan Spandau; Alireza Ghaffarieh; Yang Sun
Journal:  Sci Rep       Date:  2017-05-04       Impact factor: 4.379

3.  Novel pathogenic OCRL mutations and genotype-phenotype analysis of Chinese children affected by oculocerebrorenal syndrome: two cases and a literature review.

Authors:  Yu Zhang; Linxia Deng; Xiaohong Chen; Yingjie Hu; Yaxian Chen; Kang Chen; Jianhua Zhou
Journal:  BMC Med Genomics       Date:  2021-09-06       Impact factor: 3.063

  3 in total

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