Camptothecin (CPT), a monoterpene indole alkaloid (MIA), is the starting material for the synthesis of topotecan and irinotecan which are well known and clinically plant approved antitumor drugs. Here, we investigated the latest progress in the biosynthesis of MIA in Ophiorrhiza and identification of enzymes/genes implicated in CPT biosynthetic pathway. Secologanin and tryptamine are involved in the biosynthesis of MIA and quinoline. Due to lack of knowledge of its enzymes/genes involved, the biosynthesis of CPT has not been well established. Biotechnology plays an important role in the manufacture of pharmaceutical metabolites to maintain industrial production and diminish overexploitation of natural sources. Instead, new technologies that can be used to solve problems and to achieve overproduction of therapeutic secondary metabolites must be discovered economically viable approaches to the production of CPT. To this viewpoint, plant cell, tissue and organ cultures, cell suspension cultures, precursor feeding, hairy root cultures, bioreactors culture of suspensions/hairy root cultures, and metabolic engineering are reviewed as a costeffective and alternative for CPT production. The present chapter summarizes the latest procedures/approaches for the enhancement of CPT in Ophiorrhiza using plant cell and tissue culture. © Springer Nature Switzerland AG 2020.