Literature DB >> 2290481

Decreased erythrocyte and platelet phospholipids and fatty acids in juvenile neuronal ceroid-lipofuscinosis (Batten disease).

M J Bennett1, J H Galloway, I J Cartwright, W S Gillis, G P Hosking.   

Abstract

Total cellular and phospholipid fatty acids were analyzed in erythrocytes and platelets from six patients with juvenile neuronal ceroid-lipofuscinosis (Spielmeyer-Vogt, Batten disease, JNCL). The results were compared to those of age-matched controls. The amounts of total fatty acid and the phospholipid classes, phosphatidylcholine (PC), phosphatidylethanolamine (PE), and phosphatidylserine (PS) were significantly decreased in patients when related to cellular protein. The reductions in total fatty acids were 27% in erythrocytes and 50% in platelets. Erythrocyte PC reduction was 36%, PE was 44% and PS 27%. There were no major qualitative differences in the phospholipid fatty acids, suggesting that the fatty acid composition of the reduced phospholipid was normal, and that there is a generalized phospholipid deficiency in these cells in JNCL. This was not related to nutritional status. The pathogenesis of Batten disease may be related to abnormal membrane function resulting from this marked phospholipid deficiency.

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Year:  1990        PMID: 2290481     DOI: 10.1055/s-2008-1071496

Source DB:  PubMed          Journal:  Neuropediatrics        ISSN: 0174-304X            Impact factor:   1.947


  3 in total

Review 1.  Nosological significance of retinopathies in neurodegenerative disorders with emphasis on Batten disease.

Authors:  A A Hussain; J Marshall
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

2.  Juvenile neuronal ceroid-lipofuscinosis: characterization of the dyslipoproteinaemia and demonstration of membrane phospholipid and phospholipid-dependent signal transduction abnormalities in cultured skin fibroblasts.

Authors:  M J Bennett; S F Poirier; L Chern; A R Gayton; G P Hosking; N A Le; S Majumdar; H M Korchak
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

3.  Low erythrocyte plasmalogen and plasma docosahexaenoic acid (DHA) in juvenile neuronal ceroid-lipofuscinosis (JNCL).

Authors:  A Kohlschütter; B Schade; B Blömer; C Hübner
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

  3 in total

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