A human outbreak of West Nile virus (WNV) infection caused by WNV lineage 2 is ongoing in northern Italy. Analysis of six WNV genome sequences obtained from clinical specimens demonstrated similarities with strains circulating in central Europe and Greece and the presence of unique amino acid changes that identify a new viral strain. In addition, WNV lineage 1 Livenza, responsible for a large outbreak in northeastern Italy in 2012, was fully sequenced from a blood donor during this 2013 outbreak. A human outbreak of West Nile virus (WNV) infection is ongoing in northern Italy. We report here the clinical presentation, laboratory test results, and WNV full genome sequences from human cases of WNV infection which were diagnosed in August 2013 in the Veneto region. This region is one of the most affected by the outbreak. WNV genome sequences obtained from clinical specimens of infected individuals were subjected to phylogenetic analysis, in order to gain information on the origin and evolutionary history of the responsible viral strains. Cases of West Nile virus infection from the Veneto region Cases included five patients with laboratory-confirmed West Nile neuroinvasive disease (WNND) and four with West Nile fever (WNF), aged 51 to 88 years-old, who were resident in Rovigo, Padova, and Verona provinces (Figure 1). In addition, three WNV RNA-positive blood and organ donors were identified by screening in Padova, Verona, and Venice provinces and, besides the 12 confirmed cases, further possible cases are currently under investigation. Confirmed human cases of WNND have also been notified in regions neighbouring Veneto, namely Emilia Romagna and Lombardy [1].