Literature DB >> 10234611

Molecular heterogeneity of Krabbe disease.

L Fu1, K Inui, T Nishigaki, N Tatsumi, H Tsukamoto, C Kokubu, T Muramatsu, S Okada.   

Abstract

Krabbe disease (globoid cell leukodystrophy) is an autosomal recessive neurodegenerative disorder that affects both the central and peripheral nervous system due to an enzymatic defect of galactocerebrosidase (GALC). Following its cloning, many mutations in the galactocerebrosidase gene have been reported, but the correlation between phenotype and genotype was not clear in many cases. In this study we further investigated the molecular defects in another 10 patients (6 Japanese and 4 non-Japanese), using cultured skin fibroblasts, and found 10 mutations, of which 8 were novel, including a nonsense mutation (W647X) and 7 missense mutations (G43R, S52F, T262I, Y319C. W410G, R515H, T652R) in the coding region. Some phenotype-specific mutations were found but the other mutations were private. Mutations reported so far have been distributed over the whole GALC gene and it is difficult to speculate on functional domains of the GALC protein and phenotypically specific regions.

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Year:  1999        PMID: 10234611     DOI: 10.1023/a:1005449919660

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  15 in total

1.  Adult onset globoid cell leukodystrophy (Krabbe disease): analysis of galactosylceramidase cDNA from four Japanese patients.

Authors:  H Furuya; Y Kukita; S Nagano; Y Sakai; Y Yamashita; H Fukuyama; Y Inatomi; Y Saito; R Koike; S Tsuji; Y Fukumaki; K Hayashi; T Kobayashi
Journal:  Hum Genet       Date:  1997-09       Impact factor: 4.132

2.  Krabbe disease: isolation and characterization of a full-length cDNA for human galactocerebrosidase.

Authors:  N Sakai; K Inui; N Fujii; H Fukushima; J Nishimoto; I Yanagihara; Y Isegawa; A Iwamatsu; S Okada
Journal:  Biochem Biophys Res Commun       Date:  1994-01-28       Impact factor: 3.575

3.  Galactocerebrosidase from human urine: purification and partial characterization.

Authors:  Y Q Chen; D A Wenger
Journal:  Biochim Biophys Acta       Date:  1993-09-29

4.  Purification and characterization of galactocerebrosidase from human lymphocytes.

Authors:  N Sakai; K Inui; M Midorikawa; Y Okuno; S Ueda; A Iwamatsu; S Okada
Journal:  J Biochem       Date:  1994-09       Impact factor: 3.387

5.  Multiple mutations in the GALC gene in a patient with adult-onset Krabbe disease.

Authors:  P Luzi; M A Rafi; D A Wenger
Journal:  Ann Neurol       Date:  1996-07       Impact factor: 10.422

6.  Globoid cell leucodystrophy (Krabbe's disease): deficiency of galactocerebroside beta-galactosidase.

Authors:  K Suzuki; Y Suzuki
Journal:  Proc Natl Acad Sci U S A       Date:  1970-06       Impact factor: 11.205

7.  Cloning and expression of cDNA encoding human galactocerebrosidase, the enzyme deficient in globoid cell leukodystrophy.

Authors:  Y Q Chen; M A Rafi; G de Gala; D A Wenger
Journal:  Hum Mol Genet       Date:  1993-11       Impact factor: 6.150

8.  Molecular defects in Krabbe disease.

Authors:  N Tatsumi; K Inui; N Sakai; H Fukushima; J Nishimoto; I Yanagihara; T Nishigaki; H Tsukamoto; L Fu; M Taniike
Journal:  Hum Mol Genet       Date:  1995-10       Impact factor: 6.150

9.  A large deletion together with a point mutation in the GALC gene is a common mutant allele in patients with infantile Krabbe disease.

Authors:  M A Rafi; P Luzi; Y Q Chen; D A Wenger
Journal:  Hum Mol Genet       Date:  1995-08       Impact factor: 6.150

Review 10.  Late-onset Krabbe disease (globoid cell leukodystrophy): clinical and biochemical features of 15 cases.

Authors:  E H Kolodny; S Raghavan; W Krivit
Journal:  Dev Neurosci       Date:  1991       Impact factor: 2.984

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  12 in total

Review 1.  Biochemical, cell biological, pathological, and therapeutic aspects of Krabbe's disease.

Authors:  Je-Seong Won; Avtar K Singh; Inderjit Singh
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

Review 2.  Krabbe Disease in the Arab World.

Authors:  Hatem Zayed
Journal:  J Pediatr Genet       Date:  2015-03

3.  Chaperone therapy for Krabbe disease: potential for late-onset GALC mutations.

Authors:  Mohammad Arif Hossain; Katsumi Higaki; Seiji Saito; Kazuki Ohno; Hitoshi Sakuraba; Eiji Nanba; Yoshiyuki Suzuki; Keiichi Ozono; Norio Sakai
Journal:  J Hum Genet       Date:  2015-06-25       Impact factor: 3.172

4.  Insights into Krabbe disease from structures of galactocerebrosidase.

Authors:  Janet E Deane; Stephen C Graham; Nee Na Kim; Penelope E Stein; Rosamund McNair; M Begoña Cachón-González; Timothy M Cox; Randy J Read
Journal:  Proc Natl Acad Sci U S A       Date:  2011-08-29       Impact factor: 11.205

5.  Molecular characterization of mutations that cause globoid cell leukodystrophy and pharmacological rescue using small molecule chemical chaperones.

Authors:  Wing C Lee; Dongcheul Kang; Ena Causevic; Aimee R Herdt; Elizabeth A Eckman; Christopher B Eckman
Journal:  J Neurosci       Date:  2010-04-21       Impact factor: 6.167

6.  Structural snapshots illustrate the catalytic cycle of β-galactocerebrosidase, the defective enzyme in Krabbe disease.

Authors:  Chris H Hill; Stephen C Graham; Randy J Read; Janet E Deane
Journal:  Proc Natl Acad Sci U S A       Date:  2013-12-02       Impact factor: 11.205

7.  Identification and characterization of 15 novel GALC gene mutations causing Krabbe disease.

Authors:  Barbara Tappino; Roberta Biancheri; Matthew Mort; Stefano Regis; Fabio Corsolini; Andrea Rossi; Marina Stroppiano; Susanna Lualdi; Agata Fiumara; Bruno Bembi; Maja Di Rocco; David N Cooper; Mirella Filocamo
Journal:  Hum Mutat       Date:  2010-12       Impact factor: 4.878

8.  Azasugar inhibitors as pharmacological chaperones for Krabbe disease.

Authors:  Chris H Hill; Agnete H Viuff; Samantha J Spratley; Stéphane Salamone; Stig H Christensen; Randy J Read; Nigel W Moriarty; Henrik H Jensen; Janet E Deane
Journal:  Chem Sci       Date:  2015-03-30       Impact factor: 9.825

9.  Krabbe Disease: Report of a Rare Lipid Storage and Neurodegenerative Disorder.

Authors:  Pratyusha Pavuluri; Sabitha Vadakedath; Rajkumar Gundu; Sushmitha Uppulety; Venkataramana Kandi
Journal:  Cureus       Date:  2017-01-01

10.  Patient fibroblasts-derived induced neurons demonstrate autonomous neuronal defects in adult-onset Krabbe disease.

Authors:  Su Min Lim; Byung-Ok Choi; Seong-Il Oh; Won Jun Choi; Ki-Wook Oh; Minyeop Nahm; Yuanchao Xue; Jae Hyeok Choi; Ji Young Choi; Young-Eun Kim; Ki Wha Chung; Xiang-Dong Fu; Chang-Seok Ki; Seung Hyun Kim
Journal:  Oncotarget       Date:  2016-11-15
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