AI Article Synopsis

  • Krabbe disease is a genetic neurodegenerative disorder caused by the lack of β-galactocerebrosidase, leading to issues in breaking down key lipids in the brain's myelin.
  • There are two forms of the disease: an infantile form (90% of cases) and a late-onset form (10% of cases), with over 130 mutations found in the β-galactocerebrosidase gene.
  • In a study of 19 cases in Greece, 11 patients showed high levels of chitotriosidase, and several different mutations were identified, including one prevalent mutation (p.I250T), mostly seen in Greek patients.

Article 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.

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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5059214PMC
http://dx.doi.org/10.1007/8904_2015_457DOI Listing

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