Literature DB >> 25595337

A t(3;9)(q25.1;q34.3) translocation leading to OLFM1 fusion transcripts in Gilles de la Tourette syndrome, OCD and ADHD.

Birgitte Bertelsen1, Linea Melchior1, Lars Riff Jensen2, Camilla Groth3, Lusine Nazaryan1, Nanette Mol Debes3, Liselotte Skov3, Gangcai Xie4, Wei Sun4, Karen Brøndum-Nielsen1, Andreas Walter Kuss2, Wei Chen4, Zeynep Tümer5.   

Abstract

Gilles de la Tourette syndrome (GTS) is a neuropsychiatric disorder with a strong genetic etiology; however, finding of candidate genes is hampered by its genetic heterogeneity and the influence of non-genetic factors on disease pathogenesis. We report a case of a male patient with GTS, obsessive compulsive disorder, attention-deficit/hyperactivity-disorder, as well as other comorbidities, and a translocation t(3;9)(q25.1;q34.3) inherited from a mother with tics. Mate-pair sequencing revealed that the translocation breakpoints truncated the olfactomedin 1 (OLFM1) gene and two uncharacterized transcripts. Reverse-transcription PCR identified several fusion transcripts in the carriers, and OLFM1 expression was found to be high in GTS-related human brain regions. As OLFM1 plays a role in neuronal development it is a likely candidate gene for neuropsychiatric disorders and haploinsufficiency of OLFM1 could be a contributing risk factor to the phenotype of the carriers. In addition, one of the fusion transcripts may exert a dominant-negative or gain-of-function effect. OLFM1 is unlikely to be a major GTS susceptibility gene as no point mutations or copy number variants affecting OLFM1 were identified in 175 additional patients. The translocation described is thus a unique event, but further studies in larger cohorts are required to elucidate involvement of OLFM1 in GTS pathogenesis.
Copyright © 2015 Elsevier Ireland Ltd. All rights reserved.

Entities:  

Keywords:  Fusion transcripts; Microscopically balanced translocation; Neuropsychiatric disorder.; OLFM1; Tourette syndrome

Mesh:

Substances:

Year:  2014        PMID: 25595337     DOI: 10.1016/j.psychres.2014.12.028

Source DB:  PubMed          Journal:  Psychiatry Res        ISSN: 0165-1781            Impact factor:   3.222


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  8 in total

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