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Items: 16

1.

A saposin deficiency model in Drosophila: Lysosomal storage, progressive neurodegeneration and sensory physiological decline.

Hindle SJ, Hebbar S, Schwudke D, Elliott CJ, Sweeney ST.

Neurobiol Dis. 2017 Feb;98:77-87. doi: 10.1016/j.nbd.2016.11.012. Epub 2016 Nov 30.

2.

Developmental manganese neurotoxicity in rats: Cognitive deficits in allocentric and egocentric learning and memory.

Amos-Kroohs RM, Davenport LL, Atanasova N, Abdulla ZI, Skelton MR, Vorhees CV, Williams MT.

Neurotoxicol Teratol. 2017 Jan - Feb;59:16-26. doi: 10.1016/j.ntt.2016.10.005. Epub 2016 Oct 15.

PMID:
27756629
3.

Neuronopathic Gaucher disease: dysregulated mRNAs and miRNAs in brain pathogenesis and effects of pharmacologic chaperone treatment in a mouse model.

Dasgupta N, Xu YH, Li R, Peng Y, Pandey MK, Tinch SL, Liou B, Inskeep V, Zhang W, Setchell KD, Keddache M, Grabowski GA, Sun Y.

Hum Mol Genet. 2015 Dec 15;24(24):7031-48. doi: 10.1093/hmg/ddv404. Epub 2015 Sep 29.

4.

Structure of human saposin A at lysosomal pH.

Hill CH, Read RJ, Deane JE.

Acta Crystallogr F Struct Biol Commun. 2015 Jul;71(Pt 7):895-900. doi: 10.1107/S2053230X15008584. Epub 2015 Jun 27.

5.

LIMP-2 expression is critical for β-glucocerebrosidase activity and α-synuclein clearance.

Rothaug M, Zunke F, Mazzulli JR, Schweizer M, Altmeppen H, Lüllmann-Rauch R, Kallemeijn WW, Gaspar P, Aerts JM, Glatzel M, Saftig P, Krainc D, Schwake M, Blanz J.

Proc Natl Acad Sci U S A. 2014 Oct 28;111(43):15573-8. doi: 10.1073/pnas.1405700111. Epub 2014 Oct 14.

6.

Multiple pathogenic proteins implicated in neuronopathic Gaucher disease mice.

Xu YH, Xu K, Sun Y, Liou B, Quinn B, Li RH, Xue L, Zhang W, Setchell KD, Witte D, Grabowski GA.

Hum Mol Genet. 2014 Aug 1;23(15):3943-57. doi: 10.1093/hmg/ddu105. Epub 2014 Mar 5.

7.

Autophagy-lysosome pathway associated neuropathology and axonal degeneration in the brains of alpha-galactosidase A-deficient mice.

Nelson MP, Tse TE, O'Quinn DB, Percival SM, Jaimes EA, Warnock DG, Shacka JJ.

Acta Neuropathol Commun. 2014 Feb 14;2:20. doi: 10.1186/2051-5960-2-20.

8.

GPR37 and GPR37L1 are receptors for the neuroprotective and glioprotective factors prosaptide and prosaposin.

Meyer RC, Giddens MM, Schaefer SA, Hall RA.

Proc Natl Acad Sci U S A. 2013 Jun 4;110(23):9529-34. doi: 10.1073/pnas.1219004110. Epub 2013 May 20.

9.

Substrate compositional variation with tissue/region and Gba1 mutations in mouse models--implications for Gaucher disease.

Sun Y, Zhang W, Xu YH, Quinn B, Dasgupta N, Liou B, Setchell KD, Grabowski GA.

PLoS One. 2013;8(3):e57560. doi: 10.1371/journal.pone.0057560. Epub 2013 Mar 8.

10.

Tissue-specific effects of saposin A and saposin B on glycosphingolipid degradation in mutant mice.

Sun Y, Zamzow M, Ran H, Zhang W, Quinn B, Barnes S, Witte DP, Setchell KD, Williams MT, Vorhees CV, Grabowski GA.

Hum Mol Genet. 2013 Jun 15;22(12):2435-50. doi: 10.1093/hmg/ddt096. Epub 2013 Feb 27.

11.

The role of saposin C in Gaucher disease.

Tamargo RJ, Velayati A, Goldin E, Sidransky E.

Mol Genet Metab. 2012 Jul;106(3):257-63. doi: 10.1016/j.ymgme.2012.04.024. Epub 2012 May 5. Review.

12.

Animal models for Gaucher disease research.

Farfel-Becker T, Vitner EB, Futerman AH.

Dis Model Mech. 2011 Nov;4(6):746-52. doi: 10.1242/dmm.008185. Epub 2011 Oct 4. Review.

13.

Isofagomine in vivo effects in a neuronopathic Gaucher disease mouse.

Sun Y, Ran H, Liou B, Quinn B, Zamzow M, Zhang W, Bielawski J, Kitatani K, Setchell KD, Hannun YA, Grabowski GA.

PLoS One. 2011 Apr 20;6(4):e19037. doi: 10.1371/journal.pone.0019037.

14.

Accumulation and distribution of α-synuclein and ubiquitin in the CNS of Gaucher disease mouse models.

Xu YH, Sun Y, Ran H, Quinn B, Witte D, Grabowski GA.

Mol Genet Metab. 2011 Apr;102(4):436-47. doi: 10.1016/j.ymgme.2010.12.014. Epub 2010 Dec 31.

15.

Multi-system disorders of glycosphingolipid and ganglioside metabolism.

Xu YH, Barnes S, Sun Y, Grabowski GA.

J Lipid Res. 2010 Jul;51(7):1643-75. doi: 10.1194/jlr.R003996. Epub 2010 Mar 8. Review.

16.

Neuronopathic Gaucher disease in the mouse: viable combined selective saposin C deficiency and mutant glucocerebrosidase (V394L) mice with glucosylsphingosine and glucosylceramide accumulation and progressive neurological deficits.

Sun Y, Liou B, Ran H, Skelton MR, Williams MT, Vorhees CV, Kitatani K, Hannun YA, Witte DP, Xu YH, Grabowski GA.

Hum Mol Genet. 2010 Mar 15;19(6):1088-97. doi: 10.1093/hmg/ddp580. Epub 2010 Jan 4.

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