Literature DB >> 9653647

Structural organization and sequence of CLN2, the defective gene in classical late infantile neuronal ceroid lipofuscinosis.

C G Liu1, D E Sleat, R J Donnelly, P Lobel.   

Abstract

Mutations in the CLN2 gene result in classical late infantile neuronal ceroid lipofuscinosis (LINCL), a fatal childhood neurodegenerative disease. In this report, we present the complete sequence of the human CLN2 gene and define its physical relationship with two other genes that have been previously mapped to chromosome 11p15. The CLN2 gene consists of 13 exons and 12 introns and spans 6.65 kb. By S1 mapping and primer extension, the 5'-terminus of the CLN2 mRNA was mapped to 32 nucleotides upstream of the proposed initiation codon. A number of other elements were found to be located in close proximity to CLN2, including the gene encoding transcription factor TAFII30, the gene encoding intregrin-linked kinase, and an approximately 914-bp fragment that is 82% identical to antithrombin III. In addition, an EST cDNA clone that is transcribed on the strand opposite to CLN2 and that overlaps a portion of the CLN2 gene was identified. Finally, a set of primer pairs are presented for the amplification of the coding sequences, putative promoter, and splice junctions of the CLN2 gene. Taken together, this information will facilitate the molecular analysis of and genetic testing for classical LINCL.

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Year:  1998        PMID: 9653647     DOI: 10.1006/geno.1998.5328

Source DB:  PubMed          Journal:  Genomics        ISSN: 0888-7543            Impact factor:   5.736


  15 in total

1.  Upregulation of tripeptidyl-peptidase 1 by 3-hydroxy-(2,2)-dimethyl butyrate, a brain endogenous ligand of PPARα: Implications for late-infantile Batten disease therapy.

Authors:  Sudipta Chakrabarti; Sujyoti Chandra; Avik Roy; Sridevi Dasarathi; Madhuchhanda Kundu; Kalipada Pahan
Journal:  Neurobiol Dis       Date:  2019-03-28       Impact factor: 5.996

2.  Tripeptidyl-peptidase I deficiency in classical late-infantile neuronal ceroid lipofuscinosis brain tissue. Evidence for defective peptidase rather than proteinase activity.

Authors:  M J Warburton; F Bernardini
Journal:  J Inherit Metab Dis       Date:  2000-03       Impact factor: 4.982

3.  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

4.  Mutational analysis of the defective protease in classic late-infantile neuronal ceroid lipofuscinosis, a neurodegenerative lysosomal storage disorder.

Authors:  D E Sleat; R M Gin; I Sohar; K Wisniewski; S Sklower-Brooks; R K Pullarkat; D N Palmer; T J Lerner; R M Boustany; P Uldall; A N Siakotos; R J Donnelly; P Lobel
Journal:  Am J Hum Genet       Date:  1999-06       Impact factor: 11.025

5.  Proteomic Analysis of Brain and Cerebrospinal Fluid from the Three Major Forms of Neuronal Ceroid Lipofuscinosis Reveals Potential Biomarkers.

Authors:  David E Sleat; Abla Tannous; Istvan Sohar; Jennifer A Wiseman; Haiyan Zheng; Meiqian Qian; Caifeng Zhao; Winnie Xin; Rosemary Barone; Katherine B Sims; Dirk F Moore; Peter Lobel
Journal:  J Proteome Res       Date:  2017-08-28       Impact factor: 4.466

6.  A mouse model of classical late-infantile neuronal ceroid lipofuscinosis based on targeted disruption of the CLN2 gene results in a loss of tripeptidyl-peptidase I activity and progressive neurodegeneration.

Authors:  David E Sleat; Jennifer A Wiseman; Mukarram El-Banna; Kwi-Hye Kim; Qinwen Mao; Sandy Price; Shannon L Macauley; Richard L Sidman; Michael M Shen; Qi Zhao; Marco A Passini; Beverly L Davidson; Gregory R Stewart; Peter Lobel
Journal:  J Neurosci       Date:  2004-10-13       Impact factor: 6.167

7.  A critical tryptophan and Ca2+ in activation and catalysis of TPPI, the enzyme deficient in classic late-infantile neuronal ceroid lipofuscinosis.

Authors:  Salomon Kuizon; Kathleen DiMaiuta; Marius Walus; Edmund C Jenkins; Marisol Kuizon; Elizabeth Kida; Adam A Golabek; Daniel O Espinoza; Raju K Pullarkat; Mohammed A Junaid
Journal:  PLoS One       Date:  2010-08-03       Impact factor: 3.752

8.  Neuronal ceroid lipofuscinosis type CLN2: a new rationale for the construction of phenotypic subgroups based on a survey of 25 cases in South America.

Authors:  Romina Kohan; María Noelia Carabelos; Winnie Xin; Katherine Sims; Norberto Guelbert; Inés Adriana Cismondi; Patricia Pons; Graciela Irene Alonso; Mónica Troncoso; Scarlet Witting; David A Pearce; Raquel Dodelson de Kremer; Ana María Oller-Ramírez; Inés Noher de Halac
Journal:  Gene       Date:  2012-12-22       Impact factor: 3.688

9.  Lysosomal serine protease CLN2 regulates tumor necrosis factor-alpha-mediated apoptosis in a Bid-dependent manner.

Authors:  Hélène Autefage; Virginie Albinet; Virginie Garcia; Hortense Berges; Marie-Laure Nicolau; Nicole Therville; Marie-Françoise Altié; Catherine Caillaud; Thierry Levade; Nathalie Andrieu-Abadie
Journal:  J Biol Chem       Date:  2009-02-26       Impact factor: 5.157

Review 10.  The neuronal ceroid lipofuscinoses: mutations in different proteins result in similar disease.

Authors:  Jill M Weimer; Elizabeth Kriscenski-Perry; Yasser Elshatory; David A Pearce
Journal:  Neuromolecular Med       Date:  2002       Impact factor: 4.103

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