Case Report
A 25-year-old man who was a dairy farmer in Ahmednagar, Maharashtra, India, presented with a history of slowly growing, occasionally itchy lesions on both cheeks of 20 years’ duration. Most of the right cheek was covered by a well-defined, lobulated, gray-brown verrucous mass with a cerebriform surface (Figure 1). The left cheek was covered with a gray-brown infiltrated plaque surrounded by brown-tinged monomorphic papules.
Figure 1. Gray-brown verrucous mass at presentation.
Routine investigations were normal at presentation. Tests for purified protein derivative (tuberculin) and antibodies to human immunodeficiency virus were negative. Magnetic resonance imaging of the head showed soft tissue thickening with ulcerations involving the skin, subcutaneous tissue, and underlying facial muscles of the right cheek.
On histopathology, a hematoxylin and eosin–stained section showed hyperkeratosis, parakeratosis, pseudoepitheliomatous hyperplasia, and follicular plugs in the epidermis, as well as a mixed cellular infiltrate with Langhans giant cells and sclerotic bodies in the dermis (Figure 2). Periodic acid–Schiff and methenamine silver special stains revealed sclerotic bodies.
Figure 2. Histopathology revealed sclerotic bodies (arrows) in the dermis (H&E, original magnification ×100).
Fungal culture on Sabouraud dextrose agar at 25°C and 37°C grew olive green, rugose, velvety, leathery colonies within 48 hours, with pigmentation front and reverse (Figure 3). A panfungal polymerase chain reaction assay was positive. Direct microscopic examination of a 10% potassium hydroxide mount of the colonies showed mycelia with dematiaceous septate hyphae (Figure 4), apical branching, branching conidiophores, elliptical conidia in long chains, and pathognomonic round yeastlike bodies resembling copper pennies known as sclerotic cells (also called muriform cells and medlar bodies).1,2 The causative organism was identified as Cladosporium carrionii. A final diagnosis of chromoblastomycosis was made.
Figure 3. Fungal culture on Sabouraud dextrose agar (25°C and 37°C) grew olive green, rugose, velvety, leathery colonies within 48 hours.
Figure 4. Direct microscopic examination of a 10% potassium hydroxide mount of the colonies showed mycelia with dematiaceous septate hyphae (original magnification ×40).
After 2 months of treatment with oral itraconazole 400 mg daily, there was no notable clinical improvement and fungal elements were still seen on culture. Four treatment cycles of intravenous liposomal amphotericin B 50 mg daily (1 mg/kg daily) for 15 days followed by itraconazole 200 mg daily for another 15 days caused substantial reduction and flattening of the lesion on the right side and resolution of the lesions on the left side. Healing was accompanied by central erythema and depigmentation (Figure 5). With a suspicion of continuing C carrionii activity on the right cheek, intralesional liposomal amphotericin B 0.2 mL (in a dilution of 5 mg in 1 mL) was given weekly in the peripheral hyperpigmented raised margin, which resulted in further flattening and reduction in tissue resistance. Fungal elements were absent on repeat biopsy and culture after 4 weeks.
Figure 5. Intravenous amphotericin B and oral itraconazole resulted in substantial reduction and flattening of the lesion on the right cheek. Healing was accompanied by central erythema and depigmentation.
Six months after negative culture, further cosmetic correction of the scar on the right cheek was performed with a patterned full-thickness graft for the upper half and excision with approximation of the edges for the lower half (Figure 6). Cultures have been negative for the last 20 months; as of this writing, there has been no recurrence of lesions.
Figure 6. Further cosmetic correction of the scar on the right cheek was performed with a patterned full-thickness graft for the upper half and excision with approximation of the edges for the lower half.