| Literature DB >> 25102098 |
Hirotomo Saitsu1, Jun Tohyama2, Tom Walsh3, Mitsuhiro Kato4, Yu Kobayashi2, Ming Lee3, Yoshinori Tsurusaki1, Noriko Miyake1, Yu-Ichi Goto5, Ichizo Nishino6, Akira Ohtake7, Mary-Claire King3, Naomichi Matsumoto1.
Abstract
Recently, de novo mutations in TBL1XR1 were found in two patients with autism spectrum disorders. Here, we report on a Japanese girl presenting with West syndrome, Rett syndrome-like and autistic features. Her initial development was normal until she developed a series of spasms at 5 months of age. Electroencephalogram at 7 months showed a pattern of hypsarrhythmia, which led to a diagnosis of West syndrome. Stereotypic hand movements appeared at 8 months of age, and autistic features such as deficits in communication, hyperactivity and excitability were observed later, at 4 years and 9 months. Whole exome sequencing of the patient and her parents revealed a de novo TBL1XR1 mutation [c.209 G>A (p.Gly70Asp)] occurring at an evolutionarily conserved amino acid in an F-box-like domain. Our report expands the clinical spectrum of TBL1XR1 mutations to West syndrome with Rett-like features, together with autistic features.Entities:
Mesh:
Substances:
Year: 2014 PMID: 25102098 DOI: 10.1038/jhg.2014.71
Source DB: PubMed Journal: J Hum Genet ISSN: 1434-5161 Impact factor: 3.172