Literature DB >> 10189220

Novel mutations in African American patients with glycogen storage disease Type II. Mutations in brief no. 209. Online.

N Raben1, E Lee, L Lee, R Hirschhorn, P H Plotz.   

Abstract

The infantile form of GSD II (an inherited deficiency of the lysosomal enzyme, acid alpha-glucosidase, Pompe disease) is a severe and invariably fatal disease characterized by a rapidly progressive generalized hypotonia, hepatomegaly, and cardiomegaly. We have recently demonstrated that African American patients share a common nonsense R854X mutation in exon 18 (Becker et al., 1998). Two other mutations, D645E and M519V, have been identified in individual African American patients (Hermans et al., 1993a; Huie et al., 1994a). We describe here three novel mutations in this population group: a missense W481R in exon 10, a deletion of a T1441 in exon 10, and a splicing defect at the 5' donor site of intron 8 (IVS g+la) . The splicing defect is shared by two unrelated patients and it is linked to intragenic polymorphic sites identical to those found in patients bearing the common R854X mutation.

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Year:  1999        PMID: 10189220     DOI: 10.1002/(sici)1098-1004(1999)13:1<83::aid-humu13>3.0.co;2-2

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  5 in total

1.  Splicing mutations in glycogen-storage disease type II: evaluation of the full spectrum of mutations and their relation to patients' phenotypes.

Authors:  Stefania Zampieri; Emanuele Buratti; Silvia Dominissini; Anna Lisa Montalvo; Maria Gabriela Pittis; Bruno Bembi; Andrea Dardis
Journal:  Eur J Hum Genet       Date:  2010-12-22       Impact factor: 4.246

2.  Identification and Functional Characterization of GAA Mutations in Colombian Patients Affected by Pompe Disease.

Authors:  Mónica Yasmín Niño; Heidi Eliana Mateus; Dora Janeth Fonseca; Marian A Kroos; Sandra Yaneth Ospina; Juan Fernando Mejía; Jesús Alfredo Uribe; Arnold J J Reuser; Paul Laissue
Journal:  JIMD Rep       Date:  2012-04-19

3.  Proteasome Inhibitor Bortezomib Enhances the Activity of Multiple Mutant Forms of Lysosomal α-Glucosidase in Pompe Disease.

Authors:  Yohta Shimada; Erica Nishimura; Hiroo Hoshina; Hiroshi Kobayashi; Takashi Higuchi; Yoshikatsu Eto; Hiroyuki Ida; Toya Ohashi
Journal:  JIMD Rep       Date:  2014-09-26

Review 4.  The missing puzzle piece: splicing mutations.

Authors:  Marzena A Lewandowska
Journal:  Int J Clin Exp Pathol       Date:  2013-11-15

5.  Endolysosomal N-glycan processing is critical to attain the most active form of the enzyme acid alpha-glucosidase.

Authors:  Nithya Selvan; Nickita Mehta; Suresh Venkateswaran; Nastry Brignol; Matthew Graziano; M Osman Sheikh; Yuliya McAnany; Finn Hung; Matthew Madrid; Renee Krampetz; Nicholas Siano; Anuj Mehta; Jon Brudvig; Russell Gotschall; Jill M Weimer; Hung V Do
Journal:  J Biol Chem       Date:  2021-05-07       Impact factor: 5.157

  5 in total

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