A Case of Cheilocandidiasis

Dear Editor:

Oral candidiasis is one of the most common fungal infection in humans1. It has resurged in frequency because of increase of human immunodeficiency virus infection, obesity, diabetes mellitus, as well as increased usage of corticosteroids, immunosuppressive agents and broad-spectrum antibiotics1, 2.

A 75-year-old female presented lower lip swelling accompanied by painful erythematous ulcer with whitish creamy plaque on the lower lip for 1 month (Fig. 1A). There was no lesion on her tongue, buccal mucosa, gingiva or palate. The patient had no history of trauma. She had underlying diabetes mellitus, hypertension, and a recent history of myocardial infarction. She had been admitted to the hospital a month ago in the plastic surgery unit, under the diagnosis of lower lip cellulitis, and she was treated with triple antibiotic therapy (ceftriaxone, isepamicin, metronidazole) for 1 week. KOH test had not been performed. Bacterial culture identified Staphylococcus hominis, which could be normally found on human skin and there was no growth of fungus in Sabouraud's dextrose agar medium. Although the swelling of the lip slightly improved, lesion persisted, which prompted a referral to our department for skin biopsy. Skin biopsy showed ulceration with necrosis and numerous candida spores which could be detected by periodic acid-Schiff stain (Fig. 1B). Candida albicans were detected by tissue polymerase chain reaction (Fig. 2). Under the diagnosis of oral candidiasis, the patient was treated with antifungal agent. Fluconazole 150 mg once-weekly was used for 1 month. However, because of the patient's poor adherence, we changed to itraconazole 200 mg daily for 2 months. After the treatment, the lesion showed great improvement and she is under close follow up without any sign of recurrence (Fig. 1C). We received the patient's consent form about publishing all photographic materials.

Fig. 1
(A) A 75-year-old female presented lower lip swelling accompanied by painful erythematous ulcer with whitish creamy plaque on the lower lip for 1 month. (B) Ulceration with necrosis and numerous candida spores (periodic acid-Schiff stain, ×100). (C) After the treatment, the lesion showed great improvement and she is under close follow up without any sign of recurrence.

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Fig. 2
Polymerase chain reaction of Candida albicans. P: positive, N: negative.

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In the case of lip lesion associated with angular cheilitis or atrophic tongue, the diagnosis of Candida-associated lesion is easy1, 2, 3. However, the differential diagnosis is difficult in the erosive lip lesion of sole symptom. Also, swelling of the lips is not the characteristic clinical finding of candidiasis. Although swelling has not been considered a common sign in oral candidiasis, clinicians should remember that Candida infection is one of the causes of swelling of the lip. It should strongly be suspected especially when the patient is immunocompromised with multiple underlying conditions. This case illustrates the importance of considering candidiasis when a painful ulcer or whitish plaque and the swelling appear on the lip and the lesion does not respond to conventional therapy including systemic antibiotics. Timely evaluation could prevent the significant delays in diagnosis.