Literature DB >> 7448355

A lysosomal storage disorder in mice characterized by the accumulation of several sphingolipids.

P G Pentchev, A E Gal, A D Boothe, J Fouks, F Omodeo-Sale, R O Brady.   

Abstract

A strain of BALB/c mice with an autosomal recessive neurologic disorder has been reported previously [1, 2]. The tissues of affected animals have been further examined and the activities of varius lysosomal hydrolases and levels of sphingolipids were compared to those in control mice. There was a substantial diminution of sphingomyelinase and glucocerebrosidase activities in liver, spleen, lung, thymus, and kidney of affected mice. There was a corresponding accumulation of sphingomyelin and glucocerebroside in these tissues. The activity of several other lysosomal hydrolases was elevated. Heterozygotes did not show any of the enzymatic alterations. The brain of affected animals showed substantial accumulation of the gangliosides GM3 and GM2.

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Year:  1980        PMID: 7448355

Source DB:  PubMed          Journal:  Birth Defects Orig Artic Ser        ISSN: 0547-6844


  7 in total

1.  Therapeutic potential of cyclodextrins in the treatment of Niemann-Pick type C disease.

Authors:  Benny Liu
Journal:  Clin Lipidol       Date:  2012-06

2.  Lysosome lipid storage disorder in NCTR-BALB/c mice: spleen and lung lysosomes store unesterified cholesterol but differ in their phospholipid composition.

Authors:  C Bhuvaneswaran; M D Morris
Journal:  Mol Cell Biochem       Date:  2000-11       Impact factor: 3.396

Review 3.  Types A and B Niemann-Pick disease.

Authors:  Edward H Schuchman; Robert J Desnick
Journal:  Mol Genet Metab       Date:  2016-12-16       Impact factor: 4.797

4.  2-Hydroxypropyl-β-cyclodextrin is the active component in a triple combination formulation for treatment of Niemann-Pick C1 disease.

Authors:  Jessica Davidson; Elizabeth Molitor; Samantha Moores; Sarah E Gale; Kanagaraj Subramanian; Xuntian Jiang; Rohini Sidhu; Pamela Kell; Jesse Zhang; Hideji Fujiwara; Cristin Davidson; Paul Helquist; Bruce J Melancon; Michael Grigalunas; Gang Liu; Farbod Salahi; Olaf Wiest; Xin Xu; Forbes D Porter; Nina H Pipalia; Dana L Cruz; Edward B Holson; Jean E Schaffer; Steven U Walkley; Frederick R Maxfield; Daniel S Ory
Journal:  Biochim Biophys Acta Mol Cell Biol Lipids       Date:  2019-04-30       Impact factor: 4.698

5.  Microglia activation in Niemann-Pick disease, type C1 is amendable to therapeutic intervention.

Authors:  Antony Cougnoux; Rebecca A Drummond; Amanda L Collar; James R Iben; Alexander Salman; Harrison Westgarth; Christopher A Wassif; Niamh X Cawley; Nicole Y Farhat; Keiko Ozato; Michail S Lionakis; Forbes D Porter
Journal:  Hum Mol Genet       Date:  2018-06-15       Impact factor: 6.150

Review 6.  Lipid Involvement in Neurodegenerative Diseases of the Motor System: Insights from Lysosomal Storage Diseases.

Authors:  James C Dodge
Journal:  Front Mol Neurosci       Date:  2017-11-03       Impact factor: 5.639

7.  Evaluation of the Potential Role of Proprotein Convertase Subtilisin/Kexin Type 9 (PCSK9) in Niemann-Pick Disease, Type C1.

Authors:  Niamh X Cawley; Anna T Lyons; Daniel Abebe; Christopher A Wassif; Forbes D Porter
Journal:  Int J Mol Sci       Date:  2020-03-31       Impact factor: 5.923

  7 in total

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