Gene Symbol: synj1
Description: synaptojanin 1
Alias: fb02f11, fi15a11, nrc, wu:fb02f11, wu:fi15a11, synaptojanin-1, no optokinetic response c, slacker, slak
Species: zebrafish

Top Publications

  1. Van Epps H, Yim C, Hurley J, Brockerhoff S. Investigations of photoreceptor synaptic transmission and light adaptation in the zebrafish visual mutant nrc. Invest Ophthalmol Vis Sci. 2001;42:868-74 pubmed
    To characterize the retinal physiology of the zebrafish visual mutant no optokinetic response c (nrc) and to identify the genetic map position of the nrc mutation...
  2. Allwardt B, Lall A, Brockerhoff S, Dowling J. Synapse formation is arrested in retinal photoreceptors of the zebrafish nrc mutant. J Neurosci. 2001;21:2330-42 pubmed
    ..At 6 d post-fertilization (dpf), no optokinetic reflex could be elicited in no optokinetic response c (nrc) mutant animals under any test condition...
  3. McDermott B, Baucom J, Hudspeth A. Analysis and functional evaluation of the hair-cell transcriptome. Proc Natl Acad Sci U S A. 2007;104:11820-5 pubmed
    ..Moreover, a search for candidate genes that underlie heritable deafness identified a human ortholog of a zebrafish hair-cell gene whose map location is bracketed by the markers of a deafness locus. ..
  4. Holzhausen L, Lewis A, Cheong K, Brockerhoff S. Differential role for synaptojanin 1 in rod and cone photoreceptors. J Comp Neurol. 2009;517:633-44 pubmed publisher
    b>Synaptojanin 1 (SynJ1) is a polyphosphoinositide phosphatase involved in clathrin-mediated endocytosis in conventional synapses...
  5. Van Epps H, Hayashi M, Lucast L, Stearns G, Hurley J, De Camilli P, et al. The zebrafish nrc mutant reveals a role for the polyphosphoinositide phosphatase synaptojanin 1 in cone photoreceptor ribbon anchoring. J Neurosci. 2004;24:8641-50 pubmed
    ..b>Synaptojanin 1 (Synj1) is undetectable in nrc extracts, and biochemical activities associated with it are reduced...
  6. Trapani J, Obholzer N, Mo W, Brockerhoff S, Nicolson T. Synaptojanin1 is required for temporal fidelity of synaptic transmission in hair cells. PLoS Genet. 2009;5:e1000480 pubmed publisher an ENU mutagenesis screen for zebrafish larvae with vestibular defects, encodes the lipid phosphatase Synaptojanin 1 (Synj1)...
  7. Brockerhoff S, Dowling J, Hurley J. Zebrafish retinal mutants. Vision Res. 1998;38:1335-9 pubmed
    ..Our results indicate that mutants with defects in various processes such as photoreceptor synaptic transmission, photoreceptor adaptation and cell-type specific survival and/or function can be identified using this approach. ..
  8. George A, Hayden S, Stanton G, Brockerhoff S. Arf6 and the 5'phosphatase of synaptojanin 1 regulate autophagy in cone photoreceptors. Bioessays. 2016;38 Suppl 1:S119-35 pubmed publisher
    ..Recent work has implicated synaptojanin 1 (SynJ1) in Alzheimer's disease and Parkinson's disease, although the role of this polyphosphoinositide ..
  9. Neuhauss S. Behavioral genetic approaches to visual system development and function in zebrafish. J Neurobiol. 2003;54:148-60 pubmed
    ..Here, I summarize visually mediated behaviors of both larval and adult zebrafish and their applicability for genetic screens, and present, the approaches and results of visual behavior carried out to date. ..

More Information


  1. Lagnado L. Ribbon synapses: anchors away for a fishy tale. Curr Biol. 2005;15:R102-5 pubmed
    ..A recent study of ribbon synapses in the retina of zebrafish is an excellent example of how this approach can now be applied to a vertebrate species. ..
  2. Schebesta M, Serluca F. olig1 Expression identifies developing oligodendrocytes in zebrafish and requires hedgehog and notch signaling. Dev Dyn. 2009;238:887-98 pubmed publisher
    ..Lastly, knockdown of Olig1 in an Olig2-sensitized background did result in defects in CNS myelination, indicating a functional overlap between Olig1 and Olig2 proteins. ..
  3. George A, Hayden S, Holzhausen L, Ma E, Suzuki S, Brockerhoff S. Synaptojanin 1 is required for endolysosomal trafficking of synaptic proteins in cone photoreceptor inner segments. PLoS ONE. 2014;9:e84394 pubmed publisher
    ..In this study, we show that the polyphosphoinositide phosphatase Synaptojanin 1 (SynJ1) is critical for this process...
  4. Jia S, Muto A, Orisme W, Henson H, Parupalli C, Ju B, et al. Zebrafish Cacna1fa is required for cone photoreceptor function and synaptic ribbon formation. Hum Mol Genet. 2014;23:2981-94 pubmed publisher
    ..In addition to cloning wud, we identified synaptojanin 1 (synj1) as the defective gene in slacker (slak), a blind mutant with floating synaptic ribbons...