Literature DB >> 23542076

Molecular and clinical evaluation of Turkish patients with lysinuric protein intolerance.

Ayşegül Güzel-Ozantürk1, Rıza Köksal Ozgül, Ozlem Unal, Burcu Hişmi, Halil İbrahim Aydın, Serap Sivri, Ayşegül Tokatlı, Turgay Coşkun, Erol Aksöz, Ali Dursun.   

Abstract

Lysinuric protein intolerance is an autosomal recessive metabolic disorder caused by defective transport of the cationic amino acids lysine, arginine and ornithine in the epithelial cells of the basolateral membrane in the small intestine and renal tubules. Mutations in the solute carrier family 7, member 7, SLC7A7, gene cause this multisystemic disease with a variety of clinical symptoms such as hepatosplenomegaly, osteoporosis, hypotonia, developmental delay, pulmonary insufficiency or end-stage renal disease. In the present study, genomic structure of SLC7A7 in six Turkish patients with lysinuric protein intolerance was examined in order to detect disease causing mutations by denaturing high pressure liquid chromatography and direct sequencing. Four novel mutations were identified in SLC7A7: c.223insGTC, p.Val74_Ile75insVal; c.283insTGG, p.Glu94_Thr95insTrp; c.344_347delTTGC, p.Leu115LeufsX53; and c.1099insT, p.Ile367TyrfsX16. Clinical and biochemical findings were evaluated together with these molecular analyses.
Copyright © 2013 Elsevier B.V. All rights reserved.

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Year:  2013        PMID: 23542076     DOI: 10.1016/j.gene.2013.03.033

Source DB:  PubMed          Journal:  Gene        ISSN: 0378-1119            Impact factor:   3.688


  7 in total

1.  Lung involvement in children with lysinuric protein intolerance.

Authors:  Sarah Valimahamed-Mitha; Laureline Berteloot; Héloise Ducoin; Chris Ottolenghi; Pascale de Lonlay; Jacques de Blic
Journal:  J Inherit Metab Dis       Date:  2014-10-22       Impact factor: 4.982

Review 2.  Overview of symptoms and treatment for lysinuric protein intolerance.

Authors:  Atsuko Noguchi; Tsutomu Takahashi
Journal:  J Hum Genet       Date:  2019-06-18       Impact factor: 3.172

3.  Lysinuric Protein Intolerance Presenting with Multiple Fractures.

Authors:  Jennifer E Posey; Lindsay C Burrage; Marcus J Miller; Pengfei Liu; Matthew T Hardison; Sarah H Elsea; Qin Sun; Yaping Yang; Alecia S Willis; Alan E Schlesinger; Carlos A Bacino; Brendan H Lee
Journal:  Mol Genet Metab Rep       Date:  2014

4.  A nine-month-old-boy with Atypical Hemophagocytic Lymphohistiocytosis.

Authors:  Monia Ouederni; Monia Ben Khaled; Samia Rekaya; Ilhem Ben Fraj; Fethi Mellouli; Mohamed Bejaoui
Journal:  Mediterr J Hematol Infect Dis       Date:  2017-10-16       Impact factor: 2.576

Review 5.  Amino Acid Transport Defects in Human Inherited Metabolic Disorders.

Authors:  Raquel Yahyaoui; Javier Pérez-Frías
Journal:  Int J Mol Sci       Date:  2019-12-23       Impact factor: 5.923

6.  Immune Dysregulation Mimicking Systemic Lupus Erythematosus in a Patient With Lysinuric Protein Intolerance: Case Report and Review of the Literature.

Authors:  Josefina Longeri Contreras; Mabel A Ladino; Katherine Aránguiz; Gonzalo P Mendez; Zeynep Coban-Akdemir; Bo Yuan; Richard A Gibbs; Lindsay C Burrage; James R Lupski; Ivan K Chinn; Tiphanie P Vogel; Jordan S Orange; M Cecilia Poli
Journal:  Front Pediatr       Date:  2021-05-20       Impact factor: 3.418

7.  Inducible Slc7a7 Knockout Mouse Model Recapitulates Lysinuric Protein Intolerance Disease.

Authors:  Susanna Bodoy; Fernando Sotillo; Meritxell Espino-Guarch; Maria Pia Sperandeo; Aida Ormazabal; Antonio Zorzano; Gianfranco Sebastio; Rafael Artuch; Manuel Palacín
Journal:  Int J Mol Sci       Date:  2019-10-24       Impact factor: 5.923

  7 in total

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