Purpose: The impact of tertiary lymphoid structures (TLS) in hepatocellular carcinoma (HCC) progression is being extensively investigated. However, their presence during the early steps of human liver carcinogenesis remains unknown. We thus aimed to determine whether TLS are induced in preneoplastic/early hepatic lesions, and if they are associated with a particular immune profile. Experimental Design: A series of 127 early hepatic lesions (EHL) (low/high grade dysplastic nodules, early HCC and small and progressed HCC) was included in the study. TLS were investigated by pathological reviewing. Densities of immune cells were assessed using immunohistochemistry. A subset of lesions was microdissected and gene expression profiling was performed with a custom Nanostring panel. Results: Compared to surrounding cirrhotic nodules, EHL of all stages displayed increased densities of T cells, B cells and dendritic cells. Immature Tertiary lymphoid structures (TLS) were identified in 24% of EHL. Gene expression profiling identified a subset of EHL with elevated mRNA levels of various cytokines involved in immune cells recruitment and TLS induction. This subgroup of EHL also showed overexpression of genes related to T and B cells activation and antigen presentation as well as those related to immunosuppression and immune exhaustion. Conclusions: Local immune activation occurs in the very early steps of liver carcinogenesis, however it may not be fully efficient and paradoxically favor immune evasion and progression to full-blown HCC. These results have implications for the development of anti-HCC chemo-preventive strategies in cirrhotic patients.