Centrosomes are the major microtubule organizing centers in a large number of animal cells. They are involved in diverse cellular functions like cell division, migration, sensing and motility. Despite being identified more than 100 years ago, they did not receive much attention until recent discoveries suggesting their association with human diseases. Centrosome-related defects have been observed in several human diseases including cancers, brain disorders and ciliopathies. Researchers in the field are trying to understand the relationship between centrosomes and these diseases. Accordingly, this review provides an overview of the current knowledge regarding the role of centrosomes during ciliogenesis and neural stem cell division. The review primarily focuses on the impairment of centrosome number, organization and functioning leading to a wide range of human diseases. Finally, we discuss the scope of targeting centrosomes for therapeutic purposes. © 2020 Elsevier Ltd