Literature DB >> 31236806

Genetic testing of Mucopolysaccharidoses disease using multiplex PCR- based panels of STR markers: in silico analysis of novel mutations.

Mehdi Shafaat1, Mehrdad Hashemi2, Ahmad Majd1, Maryam Abiri3, Sirous Zeinali4,5.   

Abstract

The Mucopolysaccharidoses (MPS) are group of inherited metabolic diseases caused by the deficiency of enzymes required to degrade glycosaminoglycans (GAGs) in the lysosomes. GAGs are sulfated polysaccharides involving repeating disaccharides, uronic acid and hexosamines including chondroitin sulfate (CS), dermatan sulfate (DS), heparan sulfate (HS) and keratan sulfate (KS). Hyaluronan is excluded in terms of being non-sulfated in the GAG family. Different types of mutations have been identified as the causative agent in all types of MPS. Herein, we planned to investigate the pathogenic mutations in different types of MPS including type I (IDUA gene), IIIA (SGSH) and IIIB (NAGLU) in the eight Iranian patients. Autozygosity mapping was performed to identify the potential pathogenic variants in these 8 patients indirectly with the clinical diagnosis of MPSs. so three panels of STR (Short Tandem Repeat) markres flanking IDUA, SGSH and NAGLU genes were selected for multiplex PCR amplification. Then in each family candidate gene was sequenced to identify the pathogenic mutation. Our study showed two novel mutations c.469 T > C and c.903C > G in the IDUA gene, four recurrent mutations: c.1A > C in IDUA, c.220C > T, c.1298G > A in SGSH gene and c.457G > A in the NAGLU gene. The c.1A > C in IDUA was the most common mutation in our study. In silico analysis were performed as well to predict the pathogenicity of the novel variants.

Entities:  

Keywords:  Autozygosity mapping; Iran; Linkage; Mucopolysaccharidoses (MPS); Mutation analysis

Mesh:

Substances:

Year:  2019        PMID: 31236806     DOI: 10.1007/s11011-019-00434-z

Source DB:  PubMed          Journal:  Metab Brain Dis        ISSN: 0885-7490            Impact factor:   3.584


  51 in total

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Journal:  Proc Natl Acad Sci U S A       Date:  1996-06-11       Impact factor: 11.205

Review 2.  Identification and characterization of pharmacological chaperones to correct enzyme deficiencies in lysosomal storage disorders.

Authors:  Kenneth J Valenzano; Richie Khanna; Allan C Powe; Robert Boyd; Gary Lee; John J Flanagan; Elfrida R Benjamin
Journal:  Assay Drug Dev Technol       Date:  2011-06       Impact factor: 1.738

3.  Epidemiology of mucopolysaccharidoses.

Authors:  Shaukat A Khan; Hira Peracha; Diana Ballhausen; Alfred Wiesbauer; Marianne Rohrbach; Matthias Gautschi; Robert W Mason; Roberto Giugliani; Yasuyuki Suzuki; Kenji E Orii; Tadao Orii; Shunji Tomatsu
Journal:  Mol Genet Metab       Date:  2017-05-26       Impact factor: 4.797

4.  Mortality after hematopoietic stem cell transplantation for severe mucopolysaccharidosis type I: the 30-year University of Minnesota experience.

Authors:  Nathan J Rodgers; Alexander M Kaizer; Weston P Miller; Kyle D Rudser; Paul J Orchard; Elizabeth A Braunlin
Journal:  J Inherit Metab Dis       Date:  2017-01-04       Impact factor: 4.982

Review 5.  Can mucopolysaccharidosis type I disease severity be predicted based on a patient's genotype? A comprehensive review of the literature.

Authors:  Nancy J Terlato; Gerald F Cox
Journal:  Genet Med       Date:  2003 Jul-Aug       Impact factor: 8.822

6.  Mucopolysaccharidosis type IIIB may predominantly present with an attenuated clinical phenotype.

Authors:  Marlies J Valstar; Hennie T Bruggenwirth; Renske Olmer; Ron A Wevers; Frans W Verheijen; Ben J Poorthuis; Dicky J Halley; Frits A Wijburg
Journal:  J Inherit Metab Dis       Date:  2010-09-18       Impact factor: 4.982

7.  Childhood onset of Scheie syndrome, the attenuated form of mucopolysaccharidosis I.

Authors:  Janet A Thomas; Michael Beck; Joe T R Clarke; Gerald F Cox
Journal:  J Inherit Metab Dis       Date:  2010-06-02       Impact factor: 4.982

Review 8.  Pathogenesis and treatment of spine disease in the mucopolysaccharidoses.

Authors:  Sun H Peck; Margret L Casal; Neil R Malhotra; Can Ficicioglu; Lachlan J Smith
Journal:  Mol Genet Metab       Date:  2016-06-04       Impact factor: 4.797

9.  Progressive neurologic and somatic disease in a novel mouse model of human mucopolysaccharidosis type IIIC.

Authors:  Sara Marcó; Anna Pujol; Carles Roca; Sandra Motas; Albert Ribera; Miguel Garcia; Maria Molas; Pilar Villacampa; Cristian S Melia; Víctor Sánchez; Xavier Sánchez; Joan Bertolin; Jesús Ruberte; Virginia Haurigot; Fatima Bosch
Journal:  Dis Model Mech       Date:  2016-08-04       Impact factor: 5.758

10.  Prediction of phenotypic severity in mucopolysaccharidosis type IIIA.

Authors:  Suzan J G Knottnerus; Stephanie C M Nijmeijer; Lodewijk IJlst; Heleen Te Brinke; Naomi van Vlies; Frits A Wijburg
Journal:  Ann Neurol       Date:  2017-10-26       Impact factor: 10.422

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Authors:  Betul Çelik; Saori C Tomatsu; Shunji Tomatsu; Shaukat A Khan
Journal:  Diagnostics (Basel)       Date:  2021-02-10

2.  Mucopolysaccharidosis Type I in the Russian Federation and Other Republics of the Former Soviet Union: Molecular Genetic Analysis and Epidemiology.

Authors:  E Yu Voskoboeva; T M Bookina; A N Semyachkina; S V Mikhaylova; N D Vashakmadze; G V Baydakova; E Yu Zakharova; S I Kutsev
Journal:  Front Mol Biosci       Date:  2022-01-24
  2 in total

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