Human papillomavirus (HPV)–related carcinoma with adenoid cystic–like features is a rare, recently recognized entity restricted to the sinonasal tract. By definition, it is associated with high-risk HPV infection, particularly with HPV type 33. In most cases, tumors are composed of dual cell populations, including predominant basaloid myoepithelial cells and usually inconspicuous ductal cells. Solid components with focal cribriform or tubular patterns, abrupt keratinization within tumor nests, and squamous dysplasia of the surface epithelium are characteristics of HPV-related carcinoma with adenoid cystic–like features. The immunohistochemistry of p16 followed by high-risk HPV testing may help in the differential diagnosis. Recent studies have demonstrated that the morphologic features of this entity are more diverse than initially believed. Surgical resection is the prime alternative for treatment. According to the limited data, the prognosis of this disease may be better than that of other sinonasal carcinomas.