Figure 4

Identification of altered <italic>celsr1a</italic> function underlying the <italic>frnt</italic> phenotype.

(A) Mapping by homozygosity-by-descent indicates linkage of frnt to chromosome 4. (B) Mapping score across chromosome 4. (C) Analysis of heterogeneity across chromosome four showing a broad region of homogeneity in the population indicating linkage. (D) Fine mapping of frnt showing limited recombination and resolution of the map position along chromosome 4; white bar, area showing linkage; red hashmark, position of celsr1a; top, position (megabase, Mb) on chromosome 4, zv9 assembly (https://ensembl.org); bottom number of recombinants per meiosis (rec/me) scored. (E) Chemical mutagenesis loss-of-complementation screen to identify the gene mutation underlying the frnt phenotype. Exome sequencing of identified founders having the frnt phenotype (frnt/*), identified mutations in the celsr1a gene within the mapped interval (mh36, C1693X). (F) Identified deletions/insertions within celsr1a generated through CRISPR/Cas9 genome editing that fail to complement frnt. Recovered sequences from F1 founders; guideRNA position demarcated with overlain red bar. Of the recovered lines, allele mh104_P2027A-fs11X was retained G) Identification of transposon insertion within celsr1a in frnt.H) Schematic of celsr1a and position of identified mutations.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data

Phenotype Detail
Acknowledgments
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