Literature DB >> 16005518

Traffic accidents: molecular genetic insights into the pathogenesis of the hereditary spastic paraplegias.

Cynthia Soderblom1, Craig Blackstone.   

Abstract

The hereditary spastic paraplegias (HSPs) comprise a clinically and genetically diverse group of inherited neurological disorders in which the primary manifestation is progressive spasticity and weakness of the lower limbs. The identification of over 25 genetic loci and 11 gene products for these disorders has yielded new insights into the molecular pathways involved in the pathogenesis of HSPs. In particular, causative mutations in proteins implicated in mitochondrial function, intracellular transport and trafficking, axonal development, and myelination have been identified. In many cases, the proper intracellular trafficking and distribution of molecules and organelles are ultimately thought to be involved in HSP pathogenesis. In fact, deficits in intracellular cargo trafficking and transport are concordant with the length dependence of the distal axonopathy of upper motor neurons observed in HSP patients. Through a better understanding of the functions of the HSP gene products, novel therapeutic targets for treatment and prevention are being identified.

Entities:  

Mesh:

Substances:

Year:  2005        PMID: 16005518     DOI: 10.1016/j.pharmthera.2005.06.001

Source DB:  PubMed          Journal:  Pharmacol Ther        ISSN: 0163-7258            Impact factor:   12.310


  33 in total

Review 1.  Quality control of mitochondria: protection against neurodegeneration and ageing.

Authors:  Takashi Tatsuta; Thomas Langer
Journal:  EMBO J       Date:  2008-01-23       Impact factor: 11.598

2.  Interaction of the SPG21 protein ACP33/maspardin with the aldehyde dehydrogenase ALDH16A1.

Authors:  Michael C Hanna; Craig Blackstone
Journal:  Neurogenetics       Date:  2009-01-31       Impact factor: 2.660

3.  Unique spectrum of SPAST variants in Estonian HSP patients: presence of benign missense changes but lack of exonic rearrangements.

Authors:  Mark Braschinsky; Riin Tamm; Christian Beetz; Elena Sachez-Ferrero; Elve Raukas; Siiri-Merike Lüüs; Katrin Gross-Paju; Catherine Boillot; Federico Canzian; Andres Metspalu; Sulev Haldre
Journal:  BMC Neurol       Date:  2010-03-09       Impact factor: 2.474

4.  SPG20 protein spartin is recruited to midbodies by ESCRT-III protein Ist1 and participates in cytokinesis.

Authors:  Benoît Renvoisé; Rell L Parker; Dong Yang; Joanna C Bakowska; James H Hurley; Craig Blackstone
Journal:  Mol Biol Cell       Date:  2010-08-18       Impact factor: 4.138

5.  Novel SPG11 mutations in Asian kindreds and disruption of spatacsin function in the zebrafish.

Authors:  Laura Southgate; Dimitra Dafou; Jacqueline Hoyle; Nan Li; Esther Kinning; Peter Critchley; Andrea H Németh; Kevin Talbot; Parayil S Bindu; Sanjib Sinha; Arun B Taly; Seetharam Raghavendra; Ferenc Müller; Eamonn R Maher; Richard C Trembath
Journal:  Neurogenetics       Date:  2010-10       Impact factor: 2.660

6.  Inhibition of TFG function causes hereditary axon degeneration by impairing endoplasmic reticulum structure.

Authors:  Christian Beetz; Adam Johnson; Amber L Schuh; Seema Thakur; Rita-Eva Varga; Thomas Fothergill; Nicole Hertel; Ewa Bomba-Warczak; Holger Thiele; Gudrun Nürnberg; Janine Altmüller; Renu Saxena; Edwin R Chapman; Erik W Dent; Peter Nürnberg; Anjon Audhya
Journal:  Proc Natl Acad Sci U S A       Date:  2013-03-11       Impact factor: 11.205

7.  Autoantibodies to Non-myelin Antigens as Contributors to the Pathogenesis of Multiple Sclerosis.

Authors:  Michael C Levin; Sangmin Lee; Lidia A Gardner; Yoojin Shin; Joshua N Douglas; Chelsea Cooper
Journal:  J Clin Cell Immunol       Date:  2013-06-30

8.  Atlastin GTPases are required for Golgi apparatus and ER morphogenesis.

Authors:  Neggy Rismanchi; Cynthia Soderblom; Julia Stadler; Peng-Peng Zhu; Craig Blackstone
Journal:  Hum Mol Genet       Date:  2008-02-12       Impact factor: 6.150

9.  The hereditary spastic paraplegia proteins NIPA1, spastin and spartin are inhibitors of mammalian BMP signalling.

Authors:  Hilda T H Tsang; Thomas L Edwards; Xinnan Wang; James W Connell; Rachel J Davies; Hannah J Durrington; Cahir J O'Kane; J Paul Luzio; Evan Reid
Journal:  Hum Mol Genet       Date:  2009-07-20       Impact factor: 6.150

10.  Endogenous spartin (SPG20) is recruited to endosomes and lipid droplets and interacts with the ubiquitin E3 ligases AIP4 and AIP5.

Authors:  Thomas L Edwards; Virginia E Clowes; Hilda T H Tsang; James W Connell; Christopher M Sanderson; J Paul Luzio; Evan Reid
Journal:  Biochem J       Date:  2009-09-14       Impact factor: 3.857

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.