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Widespread RNA editing dysregulation in brains from autistic individuals.

Nature neuroscience · 2019 · Vol. 22 (1) · pp. 25-36

Abstract

Transcriptomic analyses of postmortem brains have begun to elucidate molecular abnormalities in autism spectrum disorder (ASD). However, a crucial pathway involved in synaptic development, RNA editing, has not yet been studied on a genome-wide scale. Here we profiled global patterns of adenosine-to-inosine (A-to-I) editing in a large cohort of postmortem brains of people with ASD. We observed a global bias for hypoediting in ASD brains, which was shared across brain regions and involved many synaptic genes. We show that the Fragile X proteins FMRP and FXR1P interact with RNA-editing enzymes (ADAR proteins) and modulate A-to-I editing. Furthermore, we observed convergent patterns of RNA-editing alterations in ASD and Fragile X syndrome, establishing this as a molecular link between these related diseases. Our findings, which are corroborated across multiple data sets, including dup15q (genomic duplication of 15q11.2-13.1) cases associated with intellectual disability, highlight RNA-editing dysregulation in ASD and reveal new mechanisms underlying this disorder.

Publication Types

["Journal Article", "Research Support, N.I.H., Extramural", "Research Support, Non-U.S. Gov't"]

Keywords

[]

MeSH Terms

["Adenosine Deaminase", "Autistic Disorder", "Brain", "Fragile X Messenger Ribonucleoprotein 1", "Gene Expression Profiling", "Humans", "Neurons", "RNA Editing", "RNA-Binding Proteins"]

Funding

R01 HG004659 NHGRI NIH HHS (United States)
R00 HG009530 NHGRI NIH HHS (United States)
U19 MH107367 NIMH NIH HHS (United States)
U01 HG009417 NHGRI NIH HHS (United States)
K99 HG009530 NHGRI NIH HHS (United States)
U54 HG007005 NHGRI NIH HHS (United States)
R01 HG006264 NHGRI NIH HHS (United States)
T32 HG002536 NHGRI NIH HHS (United States)
R01 MH094681 NIMH NIH HHS (United States)
R01 HD036071 NICHD NIH HHS (United States)
F32 MH114620 NIMH NIH HHS (United States)

Linked Datasets (4)

GSE107895 GSE via ncbi_elink
GEO

Widespread RNA editing dysregulation in Autism Spectrum Disorders [eCLIP-seq]

Homo sapiens
8 data files
FileTypeSize
fix_FXR_In_D501_L000_R1_001.unassigned.barcoded.fastq.gz RIP-Seq 2.9 GB
fix_FXR_In_D502_701_L000_R1_001.unassigned.barcoded.fastq.gz RIP-Seq 3.3 GB
TBN_50_S37_L003_R1_001.unassigned.barcoded.fastq.gz RIP-Seq 566.8 MB
TBN_51.R1.fastq.gz RIP-Seq 72.2 MB
TBN_54_S41_L003_R1_001.unassigned.barcoded.fastq.gz RIP-Seq 551.5 MB
TBN_55.R1.fastq.gz RIP-Seq 131.5 MB
XiaoCollab1-5079_IP.R1.fastq.gz RIP-Seq 227.3 MB
XiaoCollab2-5352_IP.R1.fastq.gz RIP-Seq 199.7 MB
GSE107867 GSE via ncbi_elink
GEO

Widespread RNA editing dysregulation in Autism Spectrum Disorders [RNA-Seq]

Homo sapiens
4 data files
FileTypeSize
carrier_sample_5212.1.fastq.gz RNA-Seq 10.9 GB
carrier_sample_5529.1.fastq.gz RNA-Seq 11.3 GB
fragileX_sample_5319.1.fastq.gz RNA-Seq 12.2 GB
fragileX_sample_5746.1.fastq.gz RNA-Seq 10.8 GB
GSE107896 GSE via ncbi_elink
GEO

Widespread RNA editing dysregulation in Autism Spectrum Disorders

Homo sapiens
GSE117776 GSE via ncbi_elink
GEO

Widespread RNA editing dysregulation in Autism Spectrum Disorders II

Homo sapiens
7 data files
FileTypeSize
control_sample_103710XX.1.fastq RNA-Seq 3.1 GB
control_sample_103710XX.1.fastq RNA-Seq 3.1 GB
control_sample_UCD1407.1.fastq RNA-Seq 3.2 GB
control_sample_UCD1407.2.fastq RNA-Seq 3.2 GB
fragileX_sample_103108GP.1.fastq RNA-Seq 4.8 GB
fragileX_sample_103108GP.2.fastq RNA-Seq 4.8 GB
fragileX_sample_JS03.1.fastq RNA-Seq 3.9 GB

Potentially Related Datasets (2)

These accessions were text-mined from the PMC full text. They may be referenced for comparison, cited from other studies, or otherwise mentioned without being primary data for this paper.

GSE51264 GSE GEO
GEO

Conserved expression of lincRNA during human and macaque prefrontal cortex development and maturation

GSE59288 GSE GEO
GEO

Disruption of human-specific synaptogenesis program in autism [RNA-seq]

Analysis Pipelines (6)

RNA-seq geo_data_processing GSE107867
eCLIP geo_data_processing GSE107895
geo_data_processing GSE107896
RNA-seq geo_data_processing GSE117776
geo_data_processing GSE51264
RNA-seq geo_data_processing GSE59288