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The search for novel antimalarial compounds has shifted in recent years from target- 878

based approaches towards whole cell ‘phenotypic’ screening. Whole cell screening 879

involves assessing diverse compound libraries for the ability of compounds to inhibit 880

parasite growth (Guiguemde et al., 2012). An advantage of this approach over target- 881

based approaches is that the discovery of antimalarial compounds does not require 882

prior knowledge of the biological target. This strategy has led to the identification of 883

numerous antimalarial compounds with diverse chemical structures (Plouffe et al., 884

2008, Gamo et al., 2010, Guiguemde et al., 2010, Duffy and Avery, 2013, Avery et al., 885

2014, Plouffe et al., 2016). 886

887

Identifying the molecular target(s) of antimalarial compounds identified in phenotypic 888

screens is not necessarily straightforward. While it is not necessary to have knowledge 889

of a compound’s chemical target prior to that compound entering the clinical pipeline, 890

knowledge of the compound target enables the development of specific biochemical 891

assays for compound/target interaction that aid in medicinal chemistry efforts to 892

improve the pharmacodynamic profile of a compound (Flannery et al., 2013). For a 893

number of compounds identified in whole cell screens as having activity against the 894

asexual stage parasite, the exposure of malaria parasites to either static or increasing 895

concentrations of the compound, typically over a period of weeks to months, has given 896

rise to resistant parasites (Rottmann et al., 2010, Jimenez-Diaz et al., 2014, Lehane et 897

al., 2014, Vaidya et al., 2014, Flannery et al., 2015, Hameed et al., 2015, Golldack et al., 898

2017, Hapuarachchi et al., 2017). Genomic techniques have been used to identify 899

mutations in the resistant parasites, thereby providing insight into mechanisms of 900

resistance and, potentially, the identity of the target. 901

902

Confirming the target of a particular antimalarial compound entails a demonstration of 903

inhibition of the activity of a particular pathway and/or protein, using one or more 904

appropriate biochemical assays. One of the challenges with regard to the screening of 905

the large number of antimalarial compounds that have emerged from whole cell 906

phenotypic screens is the adaptation of biochemical assays to a high-throughput 907

format (Flannery et al., 2013). It is with this in mind that 400 structurally diverse 908

antimalarial compounds, representing some of the most promising compounds 909

identified in phenotypic screens, were curated by the Medicine’s for Malaria Venture 910

into a set of compounds called the ‘Malaria Box’ (Spangenberg et al., 2013). The 911

Malaria Box was distributed free of charge and this has enabled researchers across the 912

world to discover the targets for some of these compounds (Lehane et al., 2014, 913

Ahyong et al., 2016, Dickerman et al., 2016, Van Voorhis et al., 2016, Golldack et al., 914

2017, Hapuarachchi et al., 2017). 915

916

The high-throughput phenotypic screening of chemical libraries against the asexual 917

blood stage malaria parasite has led to the discovery of a number of compounds with 918

mechanisms of action that are different from currently administered antimalarial drugs 919

(Rottmann et al., 2010, Jimenez-Diaz et al., 2014, Hameed et al., 2015, Golldack et al., 920

2017, Hapuarachchi et al., 2017), some of which are promising clinical candidates 921

(Jimenez-Diaz et al., 2014, Hameed et al., 2015). In terms of generating clinical 922

candidates, the asexual stage parasite high-throughput screens have been successful, 923

however, it is possible that the entirety of currently accessible compound space has 924

been explored (Guiguemde et al., 2010, Guiguemde et al., 2012, Diagana, 2015) and it 925

is likely that many of the potent drug-like compounds have their growth inhibitory 926

affect by accessing one of a limited number of targets (Diagana, 2015). These include 927

the transporter PfATP4, the lipid kinase phosphatidylinositol-4-OH kinase, the 928

pyrimidine biosynthesis enzyme dihydroorotate dehydrogenase, the respiratory chain 929

complex cytochrome bc1 and the folate recycling enzyme dihydrofolate reductase 930

(Diagana, 2015). 931

932

One drawback of the high-throughput screening systems, both target-based and whole 933

cell, is that molecules that need to be biologically activated in vivo will not be detected 934

in an in vitro screen (Flannery et al., 2013). 935

936

Ideally a compound will target multiple stages of the malaria parasite lifecycle, and so, 937

whilst clearing an asexual stage infection, also block transmission and/or remove the 938

reservoir of parasites in the liver (Leroy et al., 2014, Smith et al., 2014). High- 939

throughput screens of gametocyte-stage killing activity have uncovered compounds 940

with transmission-blocking potential (Duffy and Avery, 2013, Plouffe et al., 2016). 941

Furthermore, the development of a method to screen for compounds that kill the liver 942

stage parasite has led to the discovery of a class of compounds - the 943

imidazolopiperazines - with potent activity against liver and blood-stage parasites 944

(Meister et al., 2011). Lead optimisation of the imidazolopiperazine compound led to 945

the development of the clinical candidate KAF156 (Nagle et al., 2012) which has shown 946

promising activity against P. falciparum and P. vivax in early stage clinical trials (White 947

et al., 2016). The mechanism of action of KAF156 is yet to be determined (Kuhen et 948

al., 2014). 949

950

Many compounds that have been identified in whole-cell high-throughput screens 951

affect parasite ion homeostasis (Rottmann et al., 2010, Jimenez-Diaz et al., 2014, 952

Lehane et al., 2014, Flannery et al., 2015). These compounds are discussed further in 953

Sections 1.6. 954

955

1.5. Ion regulation in the erythrocyte and parasite