Long-read sequencing of hundreds of diverse brains provides insight into the impact of structural variation on gene expression and DNA methylation
2024

Long-read sequencing reveals how structural variations affect gene expression in the brain

Sample size: 351 publication Evidence: high

Author Information

Author(s): Billingsley Kimberley J., Meredith Melissa, Daida Kensuke, Alvarez Jerez Pilar, Negi Shloka, Malik Laksh, Genner Rylee M., Moller Abraham, Zheng Xinchang, Gibson Sophia B., Mastoras Mira, Baker Breeana, Kouam Cedric, Paquette Kimberly, Jarreau Paige, Makarious Mary B., Moore Anni, Hong Samantha, Vitale Dan, Shah Syed, Monlong Jean, Pantazis Caroline B., Asri Mobin, Shafin Kishwar, Carnevali Paolo, Marenco Stefano, Auluck Pavan, Mandal Ajeet, Miga Karen H., Rhie Arang, Reed Xylena, Ding Jinhui, Cookson Mark R., Nalls Mike, Singleton Andrew, Miller Danny E., Chaisson Mark, Timp Winston, Gibbs J. Raphael, Phillippy Adam M., Kolmogorov Mikhail, Jain Miten, Sedlazeck Fritz J., Paten Benedict, Blauwendraat Cornelis

Primary Institution: Cold Spring Harbor Laboratory

Hypothesis

Long-read sequencing can uncover the impact of structural variants on gene expression and DNA methylation in the human brain.

Conclusion

The study shows that large-scale long-read sequencing can identify complex regulatory mechanisms in the brain that were previously inaccessible.

Supporting Evidence

  • The study identified approximately 234,905 structural variants in the brain samples.
  • Locally phased assemblies covered 95% of all protein-coding genes in the human genome.
  • Quantitative trait locus analyses were applied to identify structural variants impacting gene expression.

Takeaway

Scientists used a new method to look at many brains and found important changes in genes that could help us understand brain diseases.

Methodology

The study utilized long-read sequencing with a new scalable protocol and computational pipeline on brain samples from two cohorts.

Participant Demographics

Samples were from neurologically normal controls of European and African or African admixed ancestry.

Digital Object Identifier (DOI)

10.1101/2024.12.16.628723

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