Literature DB >> 26108647

The Spectrum of Krabbe Disease in Greece: Biochemical and Molecular Findings.

Evangelia Dimitriou1, Monica Cozar2, Irene Mavridou1, Daniel Grinberg2, Lluïsa Vilageliu2, Helen Michelakakis3.   

Abstract

Krabbe disease is an autosomal recessive neurodegenerative lysosomal storage disease caused by the deficiency of β-galactocerebrosidase. This deficiency results in the impaired degradation of β-galactocerebroside, a major myelin lipid, and of galactosylsphingosine. Based on the age of onset of neurological symptoms, an infantile form (90% patients) and late-onset forms (10% patients) of the disease are recognized. Over 130 disease-causing mutations have been identified in the β-galactocerebrosidase gene. We present the biochemical and molecular findings in 19 cases of Krabbe disease, 17 of them unrelated, diagnosed in Greece over the last 30 years. β-Galactocerebrosidase activity assayed in leukocyte homogenates using either the tritium-labeled or the fluorescent substrate was diagnostic for all. Increased plasma chitotriosidase activity was found in 11/15 patients.Mutational analysis, carried out in 11 unrelated cases, identified seven different mutations, four previously described (p.I250T, c.1161+6532_polyA+9kbdel, p.K139del, p.D187V) and three novel mutations (p.D610A, c.583-1 G>C, p.W132X), and seven distinct genotypes. The most prevalent mutation was mutation p.I250T, first described in a patient of Greek origin. It accounted for 36.4% (8/22) of the mutant alleles. The second most frequent mutation was c.1161+6532_polyA+9kbdel that accounted for 22.7% (5/22) of the mutant alleles. The observed frequency was lower than that described in Northern European countries and closer to that described in Italian patients.

Entities:  

Year:  2015        PMID: 26108647      PMCID: PMC5059214          DOI: 10.1007/8904_2015_457

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  32 in total

1.  Six novel mutations detected in the GALC gene in 17 Japanese patients with Krabbe disease, and new genotype-phenotype correlation.

Authors:  Chengzhe Xu; Norio Sakai; Masako Taniike; Koji Inui; Keiichi Ozono
Journal:  J Hum Genet       Date:  2006-04-11       Impact factor: 3.172

2.  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

3.  The twitcher mouse: accumulation of galactosylsphingosine and pathology of the central nervous system.

Authors:  K Tanaka; H Nagara; T Kobayashi; I Goto
Journal:  Brain Res       Date:  1989-03-20       Impact factor: 3.252

4.  Characterization of 6-hexadecanoylamino-4-methylumbelliferyl-beta-D- galactopyranoside as fluorogenic substrate of galactocerebrosidase for the diagnosis of Krabbe disease.

Authors:  G Wiederschain; S Raghavan; E Kolodny
Journal:  Clin Chim Acta       Date:  1992-01-31       Impact factor: 3.786

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

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

6.  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

7.  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

8.  Marked elevation of plasma chitotriosidase activity. A novel hallmark of Gaucher disease.

Authors:  C E Hollak; S van Weely; M H van Oers; J M Aerts
Journal:  J Clin Invest       Date:  1994-03       Impact factor: 14.808

Review 9.  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

10.  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

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  3 in total

1.  Neuroradiological, neurophysiological and molecular findings in infantile Krabbe disease: two case reports.

Authors:  E Vargiami; E Papathanasiou; S Batzios; M Kyriazi; E Dimitriou; A Anastasiou; H Michelakakis; A-K Giese; D I Zafeiriou
Journal:  Balkan J Med Genet       Date:  2016-08-02       Impact factor: 0.519

Review 2.  Immunomodulatory Effects of Chitotriosidase Enzyme.

Authors:  Mohamed A Elmonem; Lambertus P van den Heuvel; Elena N Levtchenko
Journal:  Enzyme Res       Date:  2016-01-03

3.  Natural history of Krabbe disease - a nationwide study in Germany using clinical and MRI data.

Authors:  Sarah Isabel Krieg; Ingeborg Krägeloh-Mann; Samuel Groeschel; Stefanie Beck-Wödl; Ralf A Husain; Ludger Schöls; Christiane Kehrer
Journal:  Orphanet J Rare Dis       Date:  2020-09-10       Impact factor: 4.123

  3 in total

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