(A) Schematic representation for the strategy used to generate a MEDLE2 KO line, in which the entire locus of MEDLE2 is replaced with a nanoluciferase reporter gene (Nluc) and the neomycin phosphotransferase (Neo) selection marker fused to a 2A peptide, and tdTomato, such that the parasites express a red fluorescent protein in their cytoplasm. The solid black arrow indicates the position of the Cas9-induced double-stranded break in the middle of the gene. Note that this is a different guide from the one used for C-terminal tagging. (B) PCR mapping modification of the MEDLE2 locus using genomic DNA from wild type (WT) and transgenic (MEDLE2 KO) sporozoites using the primer pairs shown in (A) and the thymidine kinase (TK) gene as a control. Note the persistence of a WT band (1392 bp) in the 5′–3′ amplification product, despite the presence of the transgene (3524 bp). (C) HCT-8 cultures were fixed 24 hr after being infected with MEDLE2 KO transgenic parasites. MEDLE2 KO parasites exhibit red fluorescence in their cytoplasm as expected (red, tdTomato, parasite cytoplasm; green, parasites VVL; blue, Hoechst). (D) The full gene PCR products from WT (1392 bp) and MEDLE2 KO parasites (3524 bp) were used for restriction digest with ScaI. A single ScaI restriction site is found in the C terminus of WT MEDLE2; however, integration of the repair cassette disrupts this site. ScaI digested WT PCR product results in two digest products: 331 bp and 1061 bp. Undigested MEDLE2 KO full gene product has the expected 3524 bp fragment, as well as a persisting 1392 bp WT band. ScaI digested MEDLE2 KO shows the 3525 bp repair cassette resistant to ScaI digest, as well as the 331 bp and 1061 bp fragments produced from digest of the unmodified MEDLE2 locus. As a result, there are multiple copies of MEDLE2 in the genome and we have only targeted one for knockout.