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A Case of Cheilocandidosis Involving the Upper and Lower Lips

Abstract



Keywords



Candidiasis Cheilitis Cheilocandidosis



Candidiasis is one of the most common fungal infections affecting the oral cavity, impacting a significant percentage of the general population. Occasionally, the diagnosis of Candida-associated lesions can be challenging due to the variety of clinical manifestations. The well-known manifest- ations of Candida-associated lesions in the oral cavity are categorized as acute pseudomembranous candidiasis, acute atrophic candidiasis, chronic hyperplastic candidiasis, chronic atrophic candidiasis, median rhomboid glossitis, and angular cheilitis. However, limited reports have been available on Candida-associated lip lesions, apart from angular cheilitis1.

A 20-year-old man presented with yellow- to white-colored plaques on his upper and lower lips for three months (Fig. 1A). No other lesions were noted on his oral cavity. He was not immunocompromised and had no underlying diseases except acne vulgaris, which was treated with oral isotre- tinoin. No evidence of recent infections, other dermatoses, or relevant environmental exposure was suspected. Laboratory tests, including a complete blood count; liver, renal, and thyroid function tests; and HIV tests, were all within normal limits. The plaques developed one month after the admin- istration of oral isotretinoin and persisted for three months without any symptoms. Initially, fungal infection was not suspected; hence, fungal culture and polymerase chain reaction (PCR) were not performed. A skin biopsy was per- formed on the lower lip to confirm the diagnosis. The punch biopsy revealed hyperkeratotic debris containing numerous fungal hyphae and spores, identifiable through periodic acid-Schiff staining (Fig. 2). Despite the absence of fungal culture results, the histopathologic detection of hyphae and spores corresponded with Candida species, leading to the diagnosis of cheilocandidosis. Terbinafine 125 mg was administered twice daily, accompanied by topical flutrimazole cream for three months. After the treatment, the lesions completely disappeared (Fig. 1B).

Diagnosing oral candidiasis with typical symptoms is not difficult for experienced dermatologists. However, atypical variants, which do not fit into the traditional categories, complicate the diagnostic process. In a case series of cheilo- candidosis, four out of five patients exhibited concurrent angular cheilitis, unlike our patient who had no additional lesions2. Given that previous literature on cheilocandidosis has exclusively reported lesions on the lower lip, the occur- rence of lesions on both the upper and lower lips in a healthy individual, without any accompanying symptoms, posed a diagnostic challenge in our case. One limitation of this case is our sole reliance on biopsy results and failure to accurately diagnose and treat Candida infection through PCR and fungal culture. Although retinoids have recently shown potential antifungal effects, it is paradoxical that our patient developed cheilocandidosis after isotretinoin treatment. Skin fragility is a known side effect of isotretinoin, and oral candidiasis has also been reported as a side effect3. Mucosal disruption, a risk factor for oral candidiasis, may be attributed to the dryness of the mucous membranes, which disrupts the natural microbial balance and colonization resistance4. Similarly, reports have shown that retinoid exposure can lead to vulvovaginal can- didiasis5.

While uncommon, our case highlights that cheilocandidosis can manifest as isolated lip lesions without any other symptoms in young adults. Cheilocandidosis could be considered a possible differential diagnosis in cases that fail to respond to conventional cheilitis treatments.

Figure 1. Yellow- to white-colored plaques on the upper and lower lips in a 20-year-old man (A) Before treatment (B) After treatment with systemic terbinafine and topical flutrimazole
Figure 2. Histologic findings of lower lip biopsy: Hyperkeratotic debris containing numerous fungal hyphae and spores (A) Hematoxylin and eosin stain, ×100 (B) Periodic acid-Schiff stain, ×200


References


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