Literature DB >> 31901039

[Genetic study of a family of neuronal ceroid lipofuscinosis caused by a heterozygous mutation of CLN6 gene].

Tie Lou1,2, Yingzhi Huang1, Minyue Dong1.   

Abstract

OBJECTIVE: To analyze the genetic cause of a family with autosomal recessive neuronal ceroid lipofuscinoses (NCL).
METHODS: The proband was screened for mutations within the coding region of the candidate genes through high-throughput targeted sequencing. Potential causative mutations were verified by PCR and Sanger sequencing in the proband and his parents. RT-PCR and TA clone sequencing were performed to investigate whether the mRNAs were abnormally spliced.
RESULTS: The sequencing results revealed compound heterozygous mutations of CLN6:c.486+2T>C and c.486+4A>T, which were respectively inherited from his parents. RT-PCR and TA cloning sequencing suggested that the mRNAs were abnormally spliced in two forms due to both mutations.
CONCLUSIONS: The compound heterozygous mutations of CLN6:c.486+2T>C and c.486+4A>T are possibly the genetic causes of the NCL family. Detection of the novel mutation has extended mutation spectrum of CLN6.

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Year:  2019        PMID: 31901039      PMCID: PMC8800761          DOI: 10.3785/j.issn.1008-9292.2019.08.04

Source DB:  PubMed          Journal:  Zhejiang Da Xue Xue Bao Yi Xue Ban        ISSN: 1008-9292


  11 in total

1.  First Japanese variant of late infantile neuronal ceroid lipofuscinosis caused by novel CLN6 mutations.

Authors:  Ryo Sato; Takehiko Inui; Wakaba Endo; Yukimune Okubo; Yusuke Takezawa; Mai Anzai; Hiroyuki Morita; Hirotomo Saitsu; Naomichi Matsumoto; Kazuhiro Haginoya
Journal:  Brain Dev       Date:  2016-05-07       Impact factor: 1.961

2.  The Effect of Nonsense Mediated Decay on Transcriptional Activity Within the Novel β-Thalassemia Mutation HBB: c.129delT.

Authors:  Luke Forster; Rasha Mesbah Ardakani; Talal Qadah; Jill Finlayson; Reza Ghassemifar
Journal:  Hemoglobin       Date:  2015-07-24       Impact factor: 0.849

3.  [Clinical, genetic and pathological features of neuronal ceroid lipofuscinosis in 5 Chinese patients].

Authors:  S C Ren; B Q Gao; Y J Wang; X J Wu; Z X Tian; Y L Sun
Journal:  Zhonghua Yi Xue Za Zhi       Date:  2016-11-22

4.  Characterisation of early changes in ovine CLN5 and CLN6 Batten disease neural cultures for the rapid screening of therapeutics.

Authors:  Hannah L Best; Nicole J Neverman; Hollie E Wicky; Nadia L Mitchell; Beulah Leitch; Stephanie M Hughes
Journal:  Neurobiol Dis       Date:  2017-01-05       Impact factor: 5.996

5.  Novel mutations in the CLN6 gene causing a variant late infantile neuronal ceroid lipofuscinosis.

Authors:  Carla A Teixeira; Janice Espinola; Liang Huo; Johannes Kohlschütter; Dixie-Ann Persaud Sawin; Berge Minassian; Carlos J P Bessa; A Guimarães; Dietrich A Stephan; Maria Clara Sá Miranda; Marcy E MacDonald; Maria Gil Ribeiro; Rose-Mary N Boustany
Journal:  Hum Mutat       Date:  2003-05       Impact factor: 4.878

Review 6.  Beyond quality control: The role of nonsense-mediated mRNA decay (NMD) in regulating gene expression.

Authors:  Sofia Nasif; Lara Contu; Oliver Mühlemann
Journal:  Semin Cell Dev Biol       Date:  2017-09-01       Impact factor: 7.727

7.  Novel mutations in CLN6 cause late-infantile neuronal ceroid lipofuscinosis without visual impairment in two unrelated patients.

Authors:  Joseph J Chin; Babak Behnam; Mariska Davids; Prashant Sharma; Wadih M Zein; Camille Wang; Xenia Chepa-Lotrea; William Brian Gallantine; Camilo Toro; David R Adams; Cynthia J Tifft; William A Gahl; May Christine V Malicdan
Journal:  Mol Genet Metab       Date:  2018-12-03       Impact factor: 4.797

8.  The neuronal ceroid-lipofuscinoses: a historical introduction.

Authors:  Matti Haltia; Hans H Goebel
Journal:  Biochim Biophys Acta       Date:  2012-08-29

9.  CLN6 disease caused by the same mutation originating in Pakistan has varying pathology.

Authors:  Rita Guerreiro; Jose T Bras; Mariana Vieira; Varun Warrier; Shakti Agrawal; Helen Stewart; Glenn Anderson; Sara E Mole
Journal:  Eur J Paediatr Neurol       Date:  2013-06-02       Impact factor: 3.140

10.  A first CLN6 variant case of late infantile neuronal ceroid lipofuscinosis caused by a homozygous mutation in a boy from China: a case report.

Authors:  Guilian Sun; Fang Yao; Zhuoling Tian; Tianjiao Ma; Zhiliang Yang
Journal:  BMC Med Genet       Date:  2018-10-01       Impact factor: 2.103

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