Literature DB >> 16417879

Krabbe disease: severe neonatal presentation with a family history of multiple sclerosis.

Inderneel Sahai1, Hagit Baris, Virginia Kimonis, Harvey L Levy.   

Abstract

Krabbe disease, also known as globoid cell leukodystrophy, is a rare autosomal recessive disorder caused by a deficiency of a lysosomal enzyme, galactocerebrosidase. This defect prevents normal turnover of the galactolipids and results in progressive demyelination. In the infantile form, symptoms typically present at 3 to 6 months of age with subsequent neurologic deterioration. We report a case with presentation on day 7 of life and rapid progression culminating in death at 10 weeks. Galactocerebrosidase activity was absent in the leukocytes, and a 30 kb deletion in the GALC gene was found. To our knowledge, this is the earliest reported death from Krabbe disease. Several family members have multiple sclerosis, which is also a demyelinating disorder. We propose that the neonatal expression could be an example of complementary gene interaction in which coinheritance of a predisposition to multiple sclerosis led to the unusual early manifestation and rapid course of Krabbe disease in this infant.

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Year:  2005        PMID: 16417879     DOI: 10.1177/08830738050200100901

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  4 in total

Review 1.  Lysosomal storage disorders in the newborn.

Authors:  Orna Staretz-Chacham; Tess C Lang; Mary E LaMarca; Donna Krasnewich; Ellen Sidransky
Journal:  Pediatrics       Date:  2009-04       Impact factor: 7.124

Review 2.  Neonatal neuroimaging findings in inborn errors of metabolism.

Authors:  Andrea Poretti; Susan I Blaser; Maarten H Lequin; Ali Fatemi; Avner Meoded; Frances J Northington; Eugen Boltshauser; Thierry A G M Huisman
Journal:  J Magn Reson Imaging       Date:  2012-05-07       Impact factor: 4.813

Review 3.  Heterozygous carriers of galactocerebrosidase mutations that cause Krabbe disease have impaired microglial function and defective repair of myelin damage.

Authors:  Nicole J Scott-Hewitt; Christopher J Folts; Mark D Noble
Journal:  Neural Regen Res       Date:  2018-03       Impact factor: 5.135

Review 4.  Clinical and neuroimaging features as diagnostic guides in neonatal neurology diseases with cerebellar involvement.

Authors:  Jessica L Klein; Monica E Lemmon; Frances J Northington; Eugen Boltshauser; Thierry A G M Huisman; Andrea Poretti
Journal:  Cerebellum Ataxias       Date:  2016-01-13
  4 in total

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