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. Author manuscript; available in PMC: 2022 Jun 21.
Published in final edited form as: Nat Rev Drug Discov. 2021 Jun 11;20(8):611–628. doi: 10.1038/s41573-021-00210-8

Table 1 |.

Drug discovery routes from zebrafish to the clinic

Drug Zebrafish screening assay Indication Status; clinical trial identifier
New applications for known targets
MEK inhibitor Prevent lymphatic disease in ARAF-mutant transgenic model Lymphatic disease Compassionate use
MAPK pathway inhibitors Rescue blood flow in BRAF-mutant transgenic model Arteriovenous malformation Compassionate use, planned clinical trials
Leflunomide Inhibit neural crest development Melanoma Phase I (on hold); NCT01611675
All-trans retinoic acid Pluripotent zebrafish blastomere culture Adenoid cystic carcinoma Phase II; NCT03999684
Olaparib plus temozolomide Adult PDX and xenograft models Rhabdomyosarcoma Phase I; NCT01858168
New target or new application
ProHema (PGE2 derivative) HSC expansion in development Leukaemia, graft versus host disease Phase II; NCT01627314, NCT00890500
Clemizole (EPX-100) and clemizole derivatives (EPX-101, EPX-102, EPX-103) Rescue of seizures in scn1lab mutant Dravet syndrome Phase II; NCT04462770
Trifluoperazine Rescue of anaemia in rsp19 mutants Diamond Blackfan anaemia Phase I; NCT03966053
New chemical entity
ORC-13661 Protection of lateral line hair cells from cisplatin and aminoglycoside Hearing loss following antibiotic treatment in patients with CF Phase II
KER-047 Dorsalization of developing embryo Fibrodysplasia ossificans progressiva; iron deficiency anaemia; iron-refractory iron deficiency anaemia Phase I; ACTRN12619000319178
PP2A activator Selective toxicity for MYC-overexpressing thymocytes T cell acute lymphoblastic leukaemia Preclinical

CF, cystic fibrosis; HSC, haematopoietic stem cell; PDX, patient-derived xenograft; PGE2, prostaglandin E2; PP2A, protein phosphatase 2A.