Literature DB >> 30209698

A novel homozygous GALC variant has been associated with Krabbe disease in a consanguineous family.

Feyza Nur Tuncer1, Sibel Aylin Ugur Iseri2, Zuhal Yapici3, Mahmut Demir4, Meryem Karaca5, Mustafa Calik6.   

Abstract

Krabbe disease (KD) or globoid cell leukodystrophy is an autosomal recessive lysosomal storage disorder involving the white matter of the peripheral and the central nervous systems. It is caused by a deficiency of galactocerebrosidase enzyme activity. The most common manifestation is the classical early onset KD that leads to patient's loss before the age of 2. Herein, we report the evaluation of a consanguineous family with three affected children manifesting severe neurological findings that ended with death before the age of 2, in an attempt to provide genetic diagnosis to the family. One of the children underwent detailed physical and neurological examinations, including brain magnetic resonance imaging (MRI) and scalp electroencephalography (EEG) evaluations. GALC genetic testing on this child enabled identification of a novel homozygous variant (NM_000153.3: c.1394C>T; p.(Thr465Ile)), which confirmed diagnosis as KD. Familial segregation of this variant was performed by PCR amplification and Sanger sequencing that revealed the parents as heterozygous carriers. We believe this novel GALC variant will not only help in genetic counseling to this family but will also aid in identification of future KD cases.

Entities:  

Keywords:  Consanguinity; GALC; Genetic analysis; Krabbe disease; Novel variation

Mesh:

Substances:

Year:  2018        PMID: 30209698     DOI: 10.1007/s10072-018-3556-2

Source DB:  PubMed          Journal:  Neurol Sci        ISSN: 1590-1874            Impact factor:   3.307


  32 in total

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Review 2.  Leukoencephalopathies and metabolic diseases.

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3.  Krabbe disease: are certain mutations disease-causing only when specific polymorphisms are present or when inherited in trans with specific second mutations?

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Journal:  Mol Genet Metab       Date:  2013-12-19       Impact factor: 4.797

4.  Expression of individual mutations and haplotypes in the galactocerebrosidase gene identified by the newborn screening program in New York State and in confirmed cases of Krabbe's disease.

Authors:  Carlos A Saavedra-Matiz; Paola Luzi; Matthew Nichols; Joseph J Orsini; Michele Caggana; David A Wenger
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

5.  Adolescent-onset Krabbe disease with an initial diagnosis of multiple sclerosis and a novel mutation.

Authors:  José Tomás; João Durães; Lúcia Lacerda; Maria Carmo Macário
Journal:  BMJ Case Rep       Date:  2015-09-22

6.  Missense mutation in mouse GALC mimics human gene defect and offers new insights into Krabbe disease.

Authors:  Gregory B Potter; Marta Santos; Muriel T Davisson; David H Rowitch; Dan L Marks; Ernesto R Bongarzone; Magdalena A Petryniak
Journal:  Hum Mol Genet       Date:  2013-04-24       Impact factor: 6.150

7.  Early infantile Krabbe disease: results of the World-Wide Krabbe Registry.

Authors:  Patricia K Duffner; Amy Barczykowski; Kabir Jalal; Li Yan; Denise M Kay; Randy L Carter
Journal:  Pediatr Neurol       Date:  2011-09       Impact factor: 3.372

8.  Late-onset Krabbe disease is predominant in Japan and its mutant precursor protein undergoes more effective processing than the infantile-onset form.

Authors:  Mohammad Arif Hossain; Takanobu Otomo; Seiji Saito; Kazuki Ohno; Hitoshi Sakuraba; Yusuke Hamada; Keiichi Ozono; Norio Sakai
Journal:  Gene       Date:  2013-11-16       Impact factor: 3.688

9.  Six novel mutations detected in the GALC gene in 17 Japanese patients with Krabbe disease, and new genotype-phenotype correlation.

Authors:  Chengzhe Xu; Norio Sakai; Masako Taniike; Koji Inui; Keiichi Ozono
Journal:  J Hum Genet       Date:  2006-04-11       Impact factor: 3.172

10.  A large deletion together with a point mutation in the GALC gene is a common mutant allele in patients with infantile Krabbe disease.

Authors:  M A Rafi; P Luzi; Y Q Chen; D A Wenger
Journal:  Hum Mol Genet       Date:  1995-08       Impact factor: 6.150

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