OSCC in patients under the age of 50 years is on the rise globally, with limited personalised treatment options. Our study uncovers a clinically actionable molecular profile in a well curated institutional cohort of OSCC patients <50 years (n=17), with validation utilising TCGA dataset (patients <50 years – n=9; patients >50 years – n=11). We demonstrate significant divergence at the genomic and transcriptomic level between these two age groups. Whole genome and transcriptomic sequencing revealed a subset of OSCC patients <50 years with EGFR amplification and increased EGFR RNA abundance. Functional assays using patient tumour-derived cell lines (PDCLs) demonstrated responsiveness to treatment with a panel of EGFR inhibitors in PDCLs with EGFR amplification. Encouragingly, afatinib, a second-generation EGFR tyrosine kinase inhibitor, proved an efficacious therapeutic opportunity. Our findings of EGFR amplification were validated with fluorescent in situ hybridisation (FISH). FISH is a widely available testing modality in diagnostic pathology laboratories, which can be readily used to identify suitable young patients for targeted therapy in the future.