Literature DB >> 7951228

Mucopolysaccharidosis type I: identification of 8 novel mutations and determination of the frequency of the two common alpha-L-iduronidase mutations (W402X and Q70X) among European patients.

S Bunge1, W J Kleijer, C Steglich, M Beck, C Zuther, C P Morris, E Schwinger, J J Hopwood, H S Scott, A Gal.   

Abstract

A group of 46 European patients with mucopolysaccharidosis type I (MPS I) was screened for mutations of the alpha-L-iduronidase gene. The 2 common nonsense mutations, W402X and Q70X, were identified in, respectively, 37% and 35% of mutant alleles. Considerable differences were seen in the frequency of these 2 mutations in patients from North Europe (Norway and Finland) and other European countries (mainly The Netherlands and Germany). In Scandinavia, W402X and Q70X account for 17% and 62% of the MPS I alleles, respectively, while in other European countries W402X is about 2.5 times more frequent (48%) than Q70X (19%). Eight novel mutations are described including 4 missense mutations, 1 nonsense mutation, 1 insertion of 2 base pairs, and 2 deletions of 1 and 12 base pairs.

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Year:  1994        PMID: 7951228     DOI: 10.1093/hmg/3.6.861

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  27 in total

1.  A system for specific, high-throughput genotyping by allele-specific primer extension on microarrays.

Authors:  T Pastinen; M Raitio; K Lindroos; P Tainola; L Peltonen; A C Syvänen
Journal:  Genome Res       Date:  2000-07       Impact factor: 9.043

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

Authors:  Mehdi Shafaat; Mehrdad Hashemi; Ahmad Majd; Maryam Abiri; Sirous Zeinali
Journal:  Metab Brain Dis       Date:  2019-06-24       Impact factor: 3.584

3.  Cumulative incidence rates of the mucopolysaccharidoses in Germany.

Authors:  F Baehner; C Schmiedeskamp; F Krummenauer; E Miebach; M Bajbouj; C Whybra; A Kohlschütter; C Kampmann; M Beck
Journal:  J Inherit Metab Dis       Date:  2005       Impact factor: 4.982

4.  The designer aminoglycoside NB84 significantly reduces glycosaminoglycan accumulation associated with MPS I-H in the Idua-W392X mouse.

Authors:  Dan Wang; Valery Belakhov; Jeyakumar Kandasamy; Timor Baasov; Su-Chen Li; Yu-Teh Li; David M Bedwell; Kim M Keeling
Journal:  Mol Genet Metab       Date:  2011-10-19       Impact factor: 4.797

5.  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

6.  A Novel Frameshift Mutation Associated with Hurler's Syndrome: A Case Report.

Authors:  Mana Kamranjam; Seyedeh Maryam Hosseini; Mohammadreza Alaei
Journal:  J Pediatr Genet       Date:  2019-04-03

7.  Enzyme replacement in a canine model of Hurler syndrome.

Authors:  R M Shull; E D Kakkis; M F McEntee; S A Kania; A J Jonas; E F Neufeld
Journal:  Proc Natl Acad Sci U S A       Date:  1994-12-20       Impact factor: 11.205

8.  Structural study on mutant alpha-L-iduronidases: insight into mucopolysaccharidosis type I.

Authors:  Kanako Sugawara; Seiji Saito; Kazuki Ohno; Torayuki Okuyama; Hitoshi Sakuraba
Journal:  J Hum Genet       Date:  2008-03-14       Impact factor: 3.172

9.  Aminoglycoside-Induced Premature Stop Codon Read-Through of Mucopolysaccharidosis Type I Patient Q70X and W402X Mutations in Cultured Cells.

Authors:  Makoto Kamei; Karissa Kasperski; Maria Fuller; Emma J Parkinson-Lawrence; Litsa Karageorgos; Valery Belakhov; Timor Baasov; John J Hopwood; Doug A Brooks
Journal:  JIMD Rep       Date:  2013-11-06

10.  Molecular genetic defect underlying alpha-L-iduronidase pseudodeficiency.

Authors:  E L Aronovich; D Pan; C B Whitley
Journal:  Am J Hum Genet       Date:  1996-01       Impact factor: 11.025

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