Literature DB >> 418756

Optic nerve in globoid leukodystrophy (Krabbe's disease). Ultrastructural changes.

S Brownstein, K Meagher-Villemure, R C Polomeno, J M Little.   

Abstract

Globoid leukodystrophy (Krabbe's disease) was diagnosed in an infant in whom a progressive neurological disorder and optic atrophy developed. At autopsy, ultrastructural examination of the optic nerves and cerebral white matter revealed characteristic tubular inclusions in globoid-epithelioid cells. Thinning of the nerve fiber and ganglion cell layers of the retina appeared to be due to retrograde degeneration of the optic nerve related to the abnormal metabolism of myelin. We discuss the pathogenesis of the clinical and pathological ocular findings, with regard to the inherited absence of the enzyme galactocerebroside beta-galactosidase and the accumulation in the optic nerve and brain of its substrates, galactocerebroside and psychosine.

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Year:  1978        PMID: 418756     DOI: 10.1001/archopht.1978.03910050466015

Source DB:  PubMed          Journal:  Arch Ophthalmol        ISSN: 0003-9950


  9 in total

Review 1.  Beyond the cherry-red spot: Ocular manifestations of sphingolipid-mediated neurodegenerative and inflammatory disorders.

Authors:  Hui Chen; Annie Y Chan; Donald U Stone; Nawajes A Mandal
Journal:  Surv Ophthalmol       Date:  2013-09-05       Impact factor: 6.048

Review 2.  Ceramide signaling in retinal degeneration.

Authors:  Hui Chen; Julie-Thu A Tran; Richard S Brush; Anisse Saadi; Abul K Rahman; Man Yu; Douglas Yasumura; Michael T Matthes; Kelly Ahern; Haidong Yang; Matthew M LaVail; Md Nawajes A Mandal
Journal:  Adv Exp Med Biol       Date:  2012       Impact factor: 2.622

3.  An Altered Sphingolipid Profile as a Risk Factor for Progressive Neurodegeneration in Long-Chain 3-Hydroxyacyl-CoA Deficiency (LCHADD).

Authors:  Sara Tucci
Journal:  Int J Mol Sci       Date:  2022-06-27       Impact factor: 6.208

4.  Thickening of the cauda equina roots: a common finding in Krabbe disease.

Authors:  Misun Hwang; Giulio Zuccoli; Ashok Panigrahy; David Rodriguez; Michele D Poe; Maria L Escolar
Journal:  Eur Radiol       Date:  2016-05-02       Impact factor: 5.315

5.  Histochemical similarities between human and animal globoid cells in Krabbe's disease: a lectin study.

Authors:  J Alroy; A A Ucci; V Goyal; A Aurilio
Journal:  Acta Neuropathol       Date:  1986       Impact factor: 17.088

Review 6.  Regulating survival and development in the retina: key roles for simple sphingolipids.

Authors:  Nora P Rotstein; Gisela E Miranda; Carolina E Abrahan; O Lorena German
Journal:  J Lipid Res       Date:  2010-01-25       Impact factor: 5.922

7.  GALC deletions increase the risk of primary open-angle glaucoma: the role of Mendelian variants in complex disease.

Authors:  Yutao Liu; Jason Gibson; Joshua Wheeler; Lydia Coulter Kwee; Cecile M Santiago-Turla; Stephen K Akafo; Paul R Lichter; Douglas E Gaasterland; Sayoko E Moroi; Pratap Challa; Leon W Herndon; Christopher A Girkin; Donald L Budenz; Julia E Richards; R Rand Allingham; Michael A Hauser
Journal:  PLoS One       Date:  2011-11-04       Impact factor: 3.240

8.  Lack of Acid Sphingomyelinase Induces Age-Related Retinal Degeneration.

Authors:  Bill X Wu; Jie Fan; Nicholas P Boyer; Russell W Jenkins; Yiannis Koutalos; Yusuf A Hannun; Craig E Crosson
Journal:  PLoS One       Date:  2015-07-13       Impact factor: 3.240

9.  Visual System Impairment in a Mouse Model of Krabbe Disease: The Twitcher Mouse.

Authors:  Ilaria Tonazzini; Chiara Cerri; Ambra Del Grosso; Sara Antonini; Manuela Allegra; Matteo Caleo; Marco Cecchini
Journal:  Biomolecules       Date:  2020-12-23
  9 in total

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