Literature DB >> 21118112

Current therapies for the soluble lysosomal forms of neuronal ceroid lipofuscinosis.

Andrew M S Wong1, Ahad A Rahim, Simon N Waddington, Jonathan D Cooper.   

Abstract

The NCLs (neuronal ceroid lipofuscinoses) are the most common inherited paediatric neurodegenerative disorder. Although genetically distinct, NCLs can be broadly divided into two categories: one in which the mutation results in a defect in a transmembrane protein, and the other where the defect lies in a soluble lysosomal enzyme. A number of therapeutic approaches are applicable to the soluble lysosomal forms of NCL based on the phenomenon of cross-correction, whereby the ubiquitously expressed mannose 6-phosphate/IGF (insulin-like growth factor) II receptor provides an avenue for endocytosis, trafficking and lysosomal processing of extracellularly delivered enzyme. The present review discusses therapeutic utilization of cross-correction by enzyme-replacement therapy, gene therapy and stem cell therapy for the NCLs, along with an overview of the recent progress in translating these treatments into the clinic.

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Year:  2010        PMID: 21118112     DOI: 10.1042/BST0381484

Source DB:  PubMed          Journal:  Biochem Soc Trans        ISSN: 0300-5127            Impact factor:   5.407


  13 in total

1.  Intravitreal enzyme replacement preserves retinal structure and function in canine CLN2 neuronal ceroid lipofuscinosis.

Authors:  Rebecca E H Whiting; Jacqueline W Pearce; Daniella P Vansteenkiste; Katherine Bibi; Stefanie Lim; Grace Robinson Kick; Leilani J Castaner; John Sinclair; Sundeep Chandra; Annalisa Nguyen; Charles A O'Neill; Martin L Katz
Journal:  Exp Eye Res       Date:  2020-07-01       Impact factor: 3.467

Review 2.  Defining and designing polymers and hydrogels for neural tissue engineering.

Authors:  Emily R Aurand; Kyle J Lampe; Kimberly B Bjugstad
Journal:  Neurosci Res       Date:  2011-12-17       Impact factor: 3.304

3.  Oral cysteamine bitartrate and N-acetylcysteine for patients with infantile neuronal ceroid lipofuscinosis: a pilot study.

Authors:  Sondra W Levin; Eva H Baker; Wadih M Zein; Zhongjian Zhang; Zenaide M N Quezado; Ning Miao; Andrea Gropman; Kurt J Griffin; Simona Bianconi; Goutam Chandra; Omar I Khan; Rafael C Caruso; Aiyi Liu; Anil B Mukherjee
Journal:  Lancet Neurol       Date:  2014-07-02       Impact factor: 44.182

4.  Assessment of disease severity in late infantile neuronal ceroid lipofuscinosis using multiparametric MR imaging.

Authors:  J P Dyke; D Sondhi; H U Voss; D C Shungu; X Mao; K Yohay; S Worgall; N R Hackett; C Hollmann; M E Yeotsas; A L Jeong; B Van de Graaf; I Cao; S M Kaminsky; L A Heier; K D Rudser; M M Souweidane; M G Kaplitt; B Kosofsky; R G Crystal; D Ballon
Journal:  AJNR Am J Neuroradiol       Date:  2012-10-04       Impact factor: 3.825

Review 5.  Progress in the Development of Small Molecule Therapeutics for the Treatment of Neuronal Ceroid Lipofuscinoses (NCLs).

Authors:  Nihar Kinarivala; Paul C Trippier
Journal:  J Med Chem       Date:  2015-11-24       Impact factor: 7.446

6.  Imaging gene delivery in a mouse model of congenital neuronal ceroid lipofuscinosis.

Authors:  L S Pike; B A Tannous; N C Deliolanis; G Hsich; D Morse; C-H Tung; M Sena-Esteves; X O Breakefield
Journal:  Gene Ther       Date:  2011-09-08       Impact factor: 5.250

7.  Sustained Neural Stem Cell-Based Intraocular Delivery of CNTF Attenuates Photoreceptor Loss in the nclf Mouse Model of Neuronal Ceroid Lipofuscinosis.

Authors:  Wanda Jankowiak; Katharina Kruszewski; Kai Flachsbarth; Christos Skevas; Gisbert Richard; Klaus Rüther; Thomas Braulke; Udo Bartsch
Journal:  PLoS One       Date:  2015-05-20       Impact factor: 3.240

8.  Rapid and Progressive Regional Brain Atrophy in CLN6 Batten Disease Affected Sheep Measured with Longitudinal Magnetic Resonance Imaging.

Authors:  Stephen J Sawiak; Sunthara Rajan Perumal; Skye R Rudiger; Loren Matthews; Nadia L Mitchell; Clive J McLaughlan; C Simon Bawden; David N Palmer; Timothy Kuchel; A Jennifer Morton
Journal:  PLoS One       Date:  2015-07-10       Impact factor: 3.240

9.  Tandem mass spectrometry assays of palmitoyl protein thioesterase 1 and tripeptidyl peptidase activity in dried blood spots for the detection of neuronal ceroid lipofuscinoses in newborns.

Authors:  Mariana Barcenas; Chang Xue; Tatyana Marushchak-Vlaskin; C Ronald Scott; Michael H Gelb; František Tureček
Journal:  Anal Chem       Date:  2014-07-21       Impact factor: 6.986

Review 10.  Clinical translation of human neural stem cells.

Authors:  Ann Tsukamoto; Nobuko Uchida; Alexandra Capela; Thorsten Gorba; Stephen Huhn
Journal:  Stem Cell Res Ther       Date:  2013-08-29       Impact factor: 6.832

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