Background: In this case report, we represent a healthy 26-year-old patient with an oral hairy leukoplakia who is HIV-seronegative. Case presentation: The histological sample showed all typical signs for oral hairy leukoplakia, including balloon cells; however, the in situ hybridisation showed a negative result for an EBV infection, which is the common sign for oral hairy leukoplakia. Conclusion: Therefore, we suggest the unusual diagnosis of "pseudo oral hairy leukoplakia" in this special case. Keywords: oral hairy leukoplakia, HIV-negative, pseudo oral hairy leukoplakia