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Citation
Wang, X., Shaw, R., Tsang, H.T.H., Reid, E., O'Kane, C.J. (2007). Drosophila spichthyin inhibits BMP signaling and regulates synaptic growth and axonal microtubules.  Nat. Neurosci. 10(2): 177--185.
FlyBase ID
FBrf0194450
Publication Type
Research paper
Abstract

To understand the functions of NIPA1, mutated in the neurodegenerative disease hereditary spastic paraplegia, and of ichthyin, mutated in autosomal recessive congenital ichthyosis, we have studied their Drosophila melanogaster ortholog, spichthyin (Spict). Spict is found on early endosomes. Loss of Spict leads to upregulation of bone morphogenetic protein (BMP) signaling and expansion of the neuromuscular junction. BMP signaling is also necessary for a normal microtubule cytoskeleton and axonal transport; analysis of loss- and gain-of-function phenotypes indicate that Spict may antagonize this function of BMP signaling. Spict interacts with BMP receptors and promotes their internalization from the plasma membrane, implying that it inhibits BMP signaling by regulating BMP receptor traffic. This is the first demonstration of a role for a hereditary spastic paraplegia protein or ichthyin family member in a specific signaling pathway, and implies disease mechanisms for hereditary spastic paraplegia that involve dependence of the microtubule cytoskeleton on BMP signaling.

PubMed ID
PubMed Central ID
PMC2464677 (PMC) (EuropePMC)
Related Publication(s)
Personal communication to FlyBase

New gene name.
O'Kane, 2005.11.18, New gene name. [FBrf0191619]

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Secondary IDs
    Language of Publication
    English
    Additional Languages of Abstract
    Parent Publication
    Publication Type
    Journal
    Abbreviation
    Nat. Neurosci.
    Title
    Nature Neuroscience
    Publication Year
    1998-
    ISBN/ISSN
    1097-6256
    Data From Reference