Super-Mendelian inheritance mediated by CRISPRCas9 in the female mouse germline, bioRxiv, 2018-07-04

AbstractA gene drive biases the transmission of a particular allele of a gene such that it is inherited at a greater frequency than by random assortment. Recently, a highly efficient gene drive was developed in insects, which leverages the sequence-targeted DNA cleavage activity of CRISPRCas9 and endogenous homology directed repair mechanisms to convert heterozygous genotypes to homozygosity. If implemented in laboratory rodents, this powerful system would enable the rapid assembly of genotypes that involve multiple genes (e.g., to model multigenic human diseases). Such complex genetic models are currently precluded by time, cost, and a requirement for a large number of animals to obtain a few individuals of the desired genotype. However, the efficiency of a CRISPRCas9 gene drive system in mammals has not yet been determined. Here, we utilize an active genetic “CopyCat” element embedded in the mouse Tyrosinase gene to detect genotype conversions after Cas9 activity in the embryo and in the germline. Although Cas9 efficiently induces double strand DNA breaks in the early embryo and is therefore highly mutagenic, these breaks are not resolved by homology directed repair. However, when Cas9 expression is limited to the developing female germline, resulting double strand breaks are resolved by homology directed repair that copies the CopyCat allele from the donor to the receiver chromosome and leads to its super-Mendelian inheritance. These results demonstrate that the CRISPRCas9 gene drive mechanism can be implemented to simplify complex genetic crosses in laboratory mice and also contribute valuable data to the ongoing debate about applications to combat invasive rodent populations in island communities.

biorxiv genetics 100-200-users 2018

Systematic assessment of GFP tag position on protein localization and growth fitness in yeast, bioRxiv, 2018-07-02

AbstractWhile protein tags are ubiquitously utilized in molecular biology, they harbor the potential to interfere with functional traits of their fusion counterparts. Systematic evaluation of the effect of protein tags on localization and function would promote accurate use of tags in experimental setups. Here we examine the effect of Green Fluorescent Protein (GFP) tagging at either the N or C terminus of budding yeast proteins on localization and functionality. We use a competition-based approach to decipher the relative fitness of two strains tagged on the same protein but on opposite termini and from that infer the correct, physiological localization for each protein and the optimal position for tagging. Our study provides a first of a kind systematic assessment of the effect of tags on the functionality of proteins and provides step towards broad investigation of protein fusion libraries.Highlights<jatslist list-type=bullet><jatslist-item>Protein tags are widely used in molecular biology although they may interfere with protein function.<jatslist-item><jatslist-item>The subcellular localization of hundreds of proteins in yeast is different when tagged at the N or the C terminus.<jatslist-item><jatslist-item>A competition based assay enables systematic deciphering of correct tagging terminus for essential proteins.<jatslist-item><jatslist-item>The presented approach can be used to derive physiologically relevant tagged libraries.<jatslist-item>

biorxiv cell-biology 200-500-users 2018

Nested oscillatory dynamics in cortical organoids model early human brain network development, bioRxiv, 2018-06-29

SUMMARYStructural and transcriptional changes during early brain maturation follow fixed developmental programs defined by genetics. However, whether this is true for functional network activity remains unknown, primarily due to experimental inaccessibility of the initial stages of the living human brain. Here, we developed cortical organoids that spontaneously display periodic and regular oscillatory network events that are dependent on glutamatergic and GABAergic signaling. These nested oscillations exhibit cross-frequency coupling, proposed to coordinate neuronal computation and communication. As evidence of potential network maturation, oscillatory activity subsequently transitioned to more spatiotemporally irregular patterns, capturing features observed in preterm human electroencephalography (EEG). These results show that the development of structured network activity in the human neocortex may follow stable genetic programming, even in the absence of external or subcortical inputs. Our approach provides novel opportunities for investigating and manipulating the role of network activity in the developing human cortex.HIGHLIGHTS<jatslist list-type=bullet><jatslist-item>Early development of human functional neural networks and oscillatory activity can be modeled in vitro.<jatslist-item><jatslist-item>Cortical organoids exhibit phase-amplitude coupling between delta oscillation (2 Hz) and high-frequency activity (100-400 Hz) during network-synchronous events.<jatslist-item><jatslist-item>Differential role of glutamate and GABA in initiating and maintaining oscillatory network activity.<jatslist-item><jatslist-item>Developmental impairment of MECP2-KO cortical organoids impacts the emergence of oscillatory activity.<jatslist-item><jatslist-item>Cortical organoid network electrophysiological signatures correlate with human preterm neonatal EEG features.<jatslist-item>eTOCBrain oscillations are a candidate mechanism for how neural populations are temporally organized to instantiate cognition and behavior. Cortical organoids initially exhibit periodic and highly regular nested oscillatory network events that eventually transition to more spatiotemporally complex activity, capturing features of late-stage preterm infant electroencephalography. Functional neural circuitry in cortical organoids exhibits emergence and development of oscillatory network dynamics similar to those found in the developing human brain.

biorxiv neuroscience 0-100-users 2018

 

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