Literature DB >> 3604683

Congenital neuronal ceroid lipofuscinosis. A case report.

I Garborg, A Torvik, J Hals, S E Tangsrud, R Lindemann.   

Abstract

A case of congenital neuronal ceroid lipofuscinosis, a 9-day-old male child of Pakistani parents, is reported. Only 5 congenital cases have been described previously. The cerebral and cerebellar cortex showed extensive nerve-cell loss. Granular material with histochemical characteristics of ceroid-lipofuscin was deposited in neurons, macrophages and glial cells throughout the brain. Similar material was found in macrophages in the lymphoid system and in certain other organs. Ultrastructurally, the material was identical with that described in the infantile type of ceroid lipofuscinosis. It is pointed out that the metabolic exchange between the blood of the mother and the foetus is not sufficient to prevent the intra-uterine progression of the disease.

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Year:  1987        PMID: 3604683     DOI: 10.1111/j.1699-0463.1987.tb00017_95a.x

Source DB:  PubMed          Journal:  Acta Pathol Microbiol Immunol Scand A        ISSN: 0108-0164


  5 in total

Review 1.  Correlations between genotype, ultrastructural morphology and clinical phenotype in the neuronal ceroid lipofuscinoses.

Authors:  Sara E Mole; Ruth E Williams; Hans H Goebel
Journal:  Neurogenetics       Date:  2005-09-28       Impact factor: 2.660

2.  Morphologic and functional correlates of synaptic pathology in the cathepsin D knockout mouse model of congenital neuronal ceroid lipofuscinosis.

Authors:  Sabine Koch; Svetlana M Molchanova; Ann K Wright; Andrew Edwards; Jonathan D Cooper; Tomi Taira; Thomas H Gillingwater; Jaana Tyynelä
Journal:  J Neuropathol Exp Neurol       Date:  2011-12       Impact factor: 3.685

3.  A mutation in the ovine cathepsin D gene causes a congenital lysosomal storage disease with profound neurodegeneration.

Authors:  J Tyynelä; I Sohar; D E Sleat; R M Gin; R J Donnelly; M Baumann; M Haltia; P Lobel
Journal:  EMBO J       Date:  2000-06-15       Impact factor: 11.598

Review 4.  Therapeutic landscape for Batten disease: current treatments and future prospects.

Authors:  Tyler B Johnson; Jacob T Cain; Katherine A White; Denia Ramirez-Montealegre; David A Pearce; Jill M Weimer
Journal:  Nat Rev Neurol       Date:  2019-03       Impact factor: 42.937

5.  Cathepsin D expression level affects alpha-synuclein processing, aggregation, and toxicity in vivo.

Authors:  Valerie Cullen; Maria Lindfors; Juliana Ng; Anders Paetau; Erika Swinton; Piotr Kolodziej; Heather Boston; Paul Saftig; John Woulfe; Mel B Feany; Liisa Myllykangas; Michael G Schlossmacher; Jaana Tyynelä
Journal:  Mol Brain       Date:  2009-02-09       Impact factor: 4.041

  5 in total

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