| Literature DB >> 29799103 |
Shuang Wu1, Yun-Lu Li1, Ning-Yi Cheng1, Chong Wang1, En-Lin Dong1, Ying-Qian Lu1, Jin-Jing Li1, Xin-Xin Guo1, Xiang Lin1, Lu-Lu Lai1, Zhi-Wei Liu1, Ning Wang2,3, Wan-Jin Chen4,5.
Abstract
Spinal muscular atrophy (SMA) is an autosomal recessive genetic disorder caused by survival motor neuron (SMN) protein deficiency leading the loss of motor neurons in the anterior horns of the spinal cord and brainstem. More than 95% of SMA patients are attributed to the homozygous deletion of survival motor neuron 1 (SMN1) gene, and approximately 5% are caused by compound heterozygous with a SMN1 deletion and a subtle mutation. Here, we identified a rare variant c.835-5T>G in intron 6 of SMN1 in a patient affected with type I SMA. We analyzed the functional consequences of this mutation on mRNA splicing in vitro. After transfecting pCI-SMN1, pCI-SMN2, and pCI-SMN1 c.835-5T>G minigenes into HEK293, Neuro-2a, and SHSY5Y cells, reverse transcription polymerase chain reaction (RT-PCR) was performed to compare the splicing effects of these minigenes. Finally, we found that this mutation resulted in the skipping of exon 7 in SMN1, which confirmed the genetic diagnosis of SMA.Entities:
Keywords: Exon skipping; Spinal muscular atrophy; Splice variant; Survival motor neuron 1
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Year: 2018 PMID: 29799103 DOI: 10.1007/s12031-018-1079-1
Source DB: PubMed Journal: J Mol Neurosci ISSN: 0895-8696 Impact factor: 3.444