Literature DB >> 17489814

A novel microdeletion in 1(p34.2p34.3), involving the SLC2A1 (GLUT1) gene, and severe delayed development.

Sascha Vermeer1, David A Koolen, Gepke Visser, Hein J L Brackel, Ineke van der Burgt, Nicole de Leeuw, Michèl A A P Willemsen, Erik A Sistermans, Rolph Pfundt, Bert B A de Vries.   

Abstract

A de novo 4.1-megabase microdeletion of chromosome 1p34.2p34.3 has been identified by array-based comparative genomic hybridization in a young male with severely delayed development, microcephaly, pronounced hypotonia, and facial dysmorphism. The deleted region encompasses 48 genes, among them the glucose transporter 1 (SLC2A1 or GLUT1) gene. The deletion of the GLUT1 gene was in line with the abnormal ratio of cerebrospinal fluid (CSF) glucose to blood glucose, indicative of GLUT1 deficiency syndrome (MIM #606777). GLUT1 deficiency syndrome is characterized by therapy-resistant infantile seizures, developmental delay, acquired microcephaly, spasticity, ataxia, and a low concentration of glucose in the CSF. It is known that a ketogenic diet can lead to better control of seizures. This case study shows that identifying a microdeletion as the cause of learning disability is not only important for genetic counselling but might also lead to therapeutic intervention.

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Year:  2007        PMID: 17489814     DOI: 10.1111/j.1469-8749.2007.00380.x

Source DB:  PubMed          Journal:  Dev Med Child Neurol        ISSN: 0012-1622            Impact factor:   5.449


  8 in total

1.  Unintended diagnosis of Von Hippel Lindau syndrome using Array Comparative Genomic Hybridization (CGH): counseling challenges arising from unexpected information.

Authors:  Jennifer Hogan; A Turner; K Tucker; L Warwick
Journal:  J Genet Couns       Date:  2012-08-16       Impact factor: 2.537

2.  Derivative chromosome 1 and GLUT1 deficiency syndrome in a sibling pair.

Authors:  Dilek Aktas; Eda G Utine; Kristin Mrasek; Anja Weise; Ferdinand von Eggeling; Kalbiye Yalaz; Nicole Posorski; Nurten Akarsu; Mehmet Alikasifoglu; Thomas Liehr; Ergul Tuncbilek
Journal:  Mol Cytogenet       Date:  2010-05-28       Impact factor: 2.009

3.  MACF1, Involved in the 1p34.2p34.3 Microdeletion Syndrome, is Essential in Cortical Progenitor Polarity and Brain Integrity.

Authors:  Minhan Ka; Jeffrey J Moffat; Woo-Yang Kim
Journal:  Cell Mol Neurobiol       Date:  2021-04-19       Impact factor: 4.231

Review 4.  Paroxysmal Movement Disorders.

Authors:  Susan Harvey; Mary D King; Kathleen M Gorman
Journal:  Front Neurol       Date:  2021-06-11       Impact factor: 4.003

5.  A de novo 1p34.2 microdeletion identifies the synaptic vesicle gene RIMS3 as a novel candidate for autism.

Authors:  Ravinesh A Kumar; Jyotsna Sudi; Timothy D Babatz; Camille W Brune; Donald Oswald; Mayon Yen; Norma J Nowak; Edwin H Cook; Susan L Christian; William B Dobyns
Journal:  J Med Genet       Date:  2009-06-21       Impact factor: 6.318

Review 6.  The clinical and genetic heterogeneity of paroxysmal dyskinesias.

Authors:  Alice R Gardiner; Fatima Jaffer; Russell C Dale; Robyn Labrum; Roberto Erro; Esther Meyer; Georgia Xiromerisiou; Maria Stamelou; Matthew Walker; Dimitri Kullmann; Tom Warner; Paul Jarman; Mike Hanna; Manju A Kurian; Kailash P Bhatia; Henry Houlden
Journal:  Brain       Date:  2015-11-23       Impact factor: 13.501

7.  Prenatal diagnosis of 1p34.3 interstitial microdeletion by aCGH in a fetus with jaw bone abnormalities.

Authors:  Themistoklis Dagklis; Elena Papageorgiou; Elisavet Siomou; Vassilis Paspaliaris; Christina Zerva; Panagiotis Chatzis; Loretta Thomaidis; Emmanouil Manolakos; Ioannis Papoulidis
Journal:  Mol Cytogenet       Date:  2016-10-06       Impact factor: 2.009

8.  Interstitial microdeletion of the 1p34.3p34.2 region.

Authors:  Joseph E Jacher; Jeffrey W Innis
Journal:  Mol Genet Genomic Med       Date:  2018-05-03       Impact factor: 2.183

  8 in total

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