Literature DB >> 15465428

Therapy of Niemann-Pick disease, type C.

Marc C Patterson1, Frances Platt.   

Abstract

Niemann-Pick disease, type C (NPC) is a progressive autosomal recessive neurodegenerative disease, characterized by late endosomal-lysosomal accumulation of multiple lipid molecules in association with abnormal tubulovesicular trafficking. The major gene product, NPC1 protein, is not suitable for transduction therapies, and gene replacement or repair is not yet practicable for NPC and related disorders. Attempts at therapy to date have focused on reduction of the accumulating molecules that are presumed to have direct or indirect toxic effects. More recent insights into the pathophysiology of NPC raise the possibility of small molecule therapies to interdict pathways triggering apoptosis and related routes to cell death and dysfunction.

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Year:  2004        PMID: 15465428     DOI: 10.1016/j.bbalip.2004.08.013

Source DB:  PubMed          Journal:  Biochim Biophys Acta        ISSN: 0006-3002


  21 in total

1.  Linear clinical progression, independent of age of onset, in Niemann-Pick disease, type C.

Authors:  Nicole M Yanjanin; Jorge I Vélez; Andrea Gropman; Kelly King; Simona E Bianconi; Sandra K Conley; Carmen C Brewer; Beth Solomon; William J Pavan; Mauricio Arcos-Burgos; Marc C Patterson; Forbes D Porter
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2010-01-05       Impact factor: 3.568

2.  Bone-marrow-derived mesenchymal stem cells promote proliferation and neuronal differentiation of Niemann-Pick type C mouse neural stem cells by upregulation and secretion of CCL2.

Authors:  Hyun Lee; Ji Eun Kang; Jong Kil Lee; Jae-Sung Bae; Hee Kyung Jin
Journal:  Hum Gene Ther       Date:  2013-06-21       Impact factor: 5.695

3.  Endocytosis of beta-cyclodextrins is responsible for cholesterol reduction in Niemann-Pick type C mutant cells.

Authors:  Anton I Rosenbaum; Guangtao Zhang; J David Warren; Frederick R Maxfield
Journal:  Proc Natl Acad Sci U S A       Date:  2010-03-08       Impact factor: 11.205

Review 4.  Cataplexy and sleep disorders in Niemann-Pick type C disease.

Authors:  Sona Nevsimalova; Vera Malinova
Journal:  Curr Neurol Neurosci Rep       Date:  2015-01       Impact factor: 5.081

Review 5.  Treatment of Niemann--pick type C disease by histone deacetylase inhibitors.

Authors:  Paul Helquist; Frederick R Maxfield; Norbert L Wiech; Olaf Wiest
Journal:  Neurotherapeutics       Date:  2013-10       Impact factor: 7.620

6.  TNF-{alpha} plays a role in hepatocyte apoptosis in Niemann-Pick type C liver disease.

Authors:  Victoria M Rimkunas; Mark J Graham; Rosanne M Crooke; Laura Liscum
Journal:  J Lipid Res       Date:  2008-09-24       Impact factor: 5.922

7.  Investigation of N-aryl-3-alkylidenepyrrolinones as potential Niemann-Pick type C disease therapeutics.

Authors:  Casey C Cosner; John T Markiewicz; Pauline Bourbon; Christopher J Mariani; Olaf Wiest; Madalina Rujoi; Anton I Rosenbaum; Amy Y Huang; Frederick R Maxfield; Paul Helquist
Journal:  J Med Chem       Date:  2009-10-22       Impact factor: 7.446

8.  Chemical screen to reduce sterol accumulation in Niemann-Pick C disease cells identifies novel lysosomal acid lipase inhibitors.

Authors:  Anton I Rosenbaum; Madalina Rujoi; Amy Y Huang; Hong Du; Gregory A Grabowski; Frederick R Maxfield
Journal:  Biochim Biophys Acta       Date:  2009-08-20

9.  Niemann-Pick C1 functions in regulating lysosomal amine content.

Authors:  Allyn M Kaufmann; Jeffrey P Krise
Journal:  J Biol Chem       Date:  2008-06-30       Impact factor: 5.157

10.  Partial blockage of sterol biosynthesis with a squalene synthase inhibitor in early postnatal Niemann-Pick type C npcnih null mice brains reduces neuronal cholesterol accumulation, abrogates astrogliosis, but may inhibit myelin maturation.

Authors:  Patrick C Reid; Song Lin; Marie T Vanier; Yoshiko Ohno-Iwashita; H James Harwood; William F Hickey; Catherine C Y Chang; Ta Yuan Chang
Journal:  J Neurosci Methods       Date:  2007-09-12       Impact factor: 2.390

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