Literature DB >> 27844444

Exome sequencing identifies a novel homozygous CLN8 mutation in a Turkish family with Northern epilepsy.

Yavuz Sahin1, Olcay Güngör2, Zeliha Gormez3, Huseyin Demirci3, Bekir Ergüner3, Gülay Güngör4, Cengiz Dilber5.   

Abstract

Neuronal ceroid lipofuscinosis (NCL), one of the most common neurodegenerative childhood-onset disorders, is characterized by autosomal-recessive inheritance, epileptic seizures, progressive psychomotor deterioration, visual impairment, and premature death. Based on the country of origin of the patients, the clinical features/courses, and the molecular genetics background of the disorder, 14 distinct NCL subtypes have been described to date. CLN8 mutation was first identified in Finnish patients, and the condition was named Northern Epilepsy (NE); however, the severe phenotype of the CLN8 gene was subsequently found outside Finland and named 'variant late-infantile' NCL. In this study, five patients and their six healthy relatives from a large Turkish consanguineous family were enrolled. The study involved detailed clinical, radiological and molecular genetic evaluations. Whole-exome sequencing and homozygosity mapping revealed a novel homozygous CLN8 mutation, c.677T>C (p.Leu226Pro). We defined NE cases in Turkey, caused by a novel mutation in CLN8. WES can be an important diagnostic method in rare cases with atypical courses.

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Keywords:  CLN8; NCL; Neuronal ceroid lipofuscinosis; Northern epilepsy

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Year:  2016        PMID: 27844444     DOI: 10.1007/s13760-016-0721-3

Source DB:  PubMed          Journal:  Acta Neurol Belg        ISSN: 0300-9009            Impact factor:   2.396


  1 in total

1.  CLN8 Mutations Presenting with a Phenotypic Continuum of Neuronal Ceroid Lipofuscinosis-Literature Review and Case Report.

Authors:  Magdalena Badura-Stronka; Anna Winczewska-Wiktor; Anna Pietrzak; Adam Sebastian Hirschfeld; Tomasz Zemojtel; Katarzyna Wołyńska; Katarzyna Bednarek-Rajewska; Monika Seget-Dubaniewicz; Agnieszka Matheisel; Anna Latos-Bielenska; Barbara Steinborn
Journal:  Genes (Basel)       Date:  2021-06-23       Impact factor: 4.096

  1 in total

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