Avian influenza A(H9N2) has been circulating in poultry across Asia, the Middle East, and Africa, posing human health risks. In Ghana, it has co-circulated among poultry with influenza A (H5N1). This report describes Ghana´s first confirmed human case of avian influenza A(H9N2) virus infection in a two-year-old boy from Upper East Region, identified through active respiratory surveillance. Molecular and genomic analyses confirmed the virus was of the G1 lineage, closely related to other West African strains, with mammalian adaptive mutations known to increase human infection potential. The child experienced mild symptoms, received outpatient care, and recovered. Health authorities conducted epidemiological investigations. No source was identified for the child´s infection; no additional human infections were detected. This case highlights the importance of robust avian influenza surveillance in animals and humans, particularly in regions with human-animal interactions. It underscores the importance of national and global collaboration using a One Health approach to detect and prevent zoonotic spillovers and potential pandemics.