PUBLICATION

zMADM (zebrafish mosaic analysis with double markers) for single-cell gene knockout and dual-lineage tracing

Authors
Xu, B., Kucenas, S., Zong, H.
ID
ZDB-PUB-220225-3
Date
2022
Source
Proceedings of the National Academy of Sciences of the United States of America   119(9): (Journal)
Registered Authors
Kucenas, Sarah
Keywords
single-cell gene knockout, lineage tracing, mosaic analysis with double markers, zebrafish
MeSH Terms
  • Animals
  • CRISPR-Cas Systems
  • Cell Lineage*
  • Gene Knockdown Techniques
  • Genetic Markers*
  • Mosaicism*
  • Single-Cell Analysis/methods*
  • Zebrafish/genetics*
PubMed
35197298 Full text @ Proc. Natl. Acad. Sci. USA
Abstract
As a vertebrate model organism, zebrafish has many unique advantages in developmental studies, regenerative biology, and disease modeling. However, tissue-specific gene knockout in zebrafish is challenging due to technical difficulties in making floxed alleles. Even when successful, tissue-level knockout can affect too many cells, making it difficult to distinguish cell autonomous from noncell autonomous gene function. Here, we present a genetic system termed zebrafish mosaic analysis with double markers (zMADM). Through Cre/loxP-mediated interchromosomal mitotic recombination of two reciprocally chimeric fluorescent genes, zMADM generates sporadic (<0.5%), GFP+ mutant cells along with RFP+ sibling wild-type cells, enabling phenotypic analysis at single-cell resolution. Using wild-type zMADM, we traced two sibling cells (GFP+ and RFP+) in real time during a dynamic developmental process. Using nf1 mutant zMADM, we demonstrated an overproliferation phenotype of nf1 mutant cells in comparison to wild-type sibling cells in the same zebrafish. The readiness of zMADM to produce sporadic mutant cells without the need to generate floxed alleles should fundamentally improve the throughput of genetic analysis in zebrafish; the lineage-tracing capability combined with phenotypic analysis at the single-cell level should lead to deep insights into developmental and disease mechanisms. Therefore, we are confident that zMADM will enable groundbreaking discoveries once broadly distributed in the field.
Genes / Markers
Figures
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping