Post-translational modifications are key molecular events that modify nascent proteins by the addition of chemical groups to selected amino acid residues. These modifications modulate their functional properties by regulating their stability, localization, interaction potential, and activity. Proteins that are destined to be embedded in the plasma membrane often undergo glycosylation or lipidation. Glycosylation describes the addition of a carbohydrate group to a protein, while lipidation describes the addition of lipid to a protein, which can help anchor that protein to the cell membrane. It is well accepted that these modifications are required for fine regulation of cell proliferation, differentiation, and apoptosis and are essential for tissue development and homeostasis. In addition, dysregulation of post-translational modifications contributes to many pathologies, including neurodegenerative diseases and deafness. A better understanding of these modifications will help to identify potential new therapeutic strategies. We aimed to focus our research on lipidation and to approach this issue, we used interdisciplinary approaches involving mouse model as well as cell biology, biochemistry, and electrophysiology.