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ABSTRACT:Oral hairy leukoplakia is a lesion that generally occurs within the tongue. It is characterized by a white patch on the side of the tongue with a corrugated or hairy appearance. The OHL affects patients with HIV infection and has characteristics that differ from oral leukoplakia. The etiology of oral hairy leukoplakia is linked to the infection of epithelial cells by Epstein-Barr virus. The objective of this paper was to report a case of atypical oral hairy leukoplakia in a patient with HIV infection. A male patient, 43 years old, was hospitalized complaining of abdominal pain and symptoms suggestive of jaundice. A white plaque was identified on his tongue during a clinical examination. That lesion was painless and not removable by scraping. A liquid-based cytology was performed and cytological analysis revealed structures suggestive of Candida sp. and cytological changes consistent with oral hairy leukoplakia (Cowdry type A inclusion bodies, and ground-glass nuclei). Exfoliative cytology is a reliable test that can be used by a dentist and otolaryngologists to determine the diagnosis of atypical cases of oral hairy leukoplakia.