 During November 2021 to May 2022, we identified 37 clinical cases of streptococcus equisub species zoopidemicus infections in central Italy. Epidemiologic investigations and whole genome sequencing showed unpasterized fresh dairy products were the outbreak source. Early diagnosis by using sequencing technology prevented the spread of life-threatening S. equisopsp. Zoopidemicus infections. This article was authored by Serena Basica, Alexandra Cavarini, Maria Elisabetta de Angelis, and others.