CEP89, also known as centrosomal protein 89, is essential for ciliogenesis, mitochondrial metabolism, and neuronal function [2]. It is involved in pathways related to the assembly and disassembly of primary cilia, as well as mitochondrial oxidative phosphorylation. CEP89 is highly conserved and ubiquitously expressed, playing a crucial role in various biological processes [2]. Genetic models, such as Drosophila and knockout cell lines, have been valuable in studying its functions.

In Drosophila, ubiquitous knockdown of fly Cep89 decreased complex IV activity and was lethal. It was also required for mitochondrial integrity, membrane depolarization, synaptic transmission in photoreceptor neurons, and (sub)synaptic organization at the larval neuromuscular junction [2]. In mammalian cells, CRISPR-Cas9 knockout studies showed that CEP89 selectively functions in RAB34+ ciliary vesicle recruitment during ciliogenesis [3]. Additionally, in ovarian cancer, more than a quarter of patients have copy number gains in the CEP89 gene. High CEP89 expression is associated with a shorter overall survival, suggesting it could be a prognostic marker and a potential therapeutic target [1].

In conclusion, CEP89 is vital for mitochondrial metabolism, especially complex IV activity, and neuronal function. Model-based research, including knockout models in flies and mammalian cells, has revealed its role in ciliary vesicle recruitment and its significance in ovarian cancer prognosis. These findings contribute to our understanding of biological processes and potential disease-related applications.