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Background Angular cheilitis, an infection mainly caused by  Candida  yeasts, is featured by the appearance of inflammatory lesions at the bilateral corners of the mouth, particularly in patients with poor oral hygiene, ill-fitting dentures and old age. The first isolation of an atypical yeast,  Cystobasidium calyptogenae,  from oral samples of a patient presenting with angular cheilitis is discussed in this study. Case presentation Angular cheilitis was diagnosed in a 60-year-old denture-wearing woman who presented with an irritation fibroma on her right lower buccal sulcus over the premolar region. Primary cultures of her oral swab and oral rinse samples grew a pure culture of an uncommon yeast strain resembling  Rhodotorula  sp. Sequence analysis of the yeast internal transcribed spacer (ITS) gene region and D1D2 domain showed highest similarity (99.6% and 100%, respectively) to  C. calyptogenae  CBS 9125 type strain. Following 2 weeks of treatment with miconazole/fusidic acid and mouthwash, the oral lesion showed improvement with less erythema.  C. calyptogenae  was not isolated from the patient’s oral samples upon repeat sampling. Conclusion This is the first report on the isolation of  C. calyptogenae  from human oral samples. The ability of  C. calyptogenae  to grow at 37 °C and the fact that it was the only yeast species isolated from the patient’s oral samples suggests its pathogenic potential and possible involvement in angular cheilitis. The ubiquitous nature of the  Cystobasidium  yeast is believed to increase the likelihood of opportunistic infections among immunocompromised individuals. As  Cystobasidium  is phenotypically indistinguishable from  Rhodotorula,  an emerging opportunistic pathogen, surveillance using molecular identification in clinical settings is essential in providing accurate diagnosis and treatment of uncommon yeast infections.

First isolation and identification of Cystobasidium calyptogenae from the oral samples of an elderly patient presenting with angular cheilitis