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Primary cutaneous trichosporonosis in immunocompetent individuals is very rare.
A woman in her 20s presented with a large erythematous indurated plaque involving the face and neck that had been first noted at age 2 years. The plaque progressively increased in size, and similar plaques developed over the back, right dorsal surface of the hand, suprapubic area (Figure 1A), and right thigh over the last 4 years. Scarring developed in the plaques over the face and hand during the course of the disease and led to flexion contracture of the hand with limitation of movement. There were no constitutional symptoms. There was no history of trauma preceding these lesions. She had received multiple modalities of treatments, including antileprosy therapy for 1.5 years, antitubercular therapy for 2 months, antileishmanial therapy with amphotericin B for 3 months, and anti-fungal therapy with itraconazole for 3 months, without any significant improvement.
A, Well-defined erythematous plaque of 7 × 8 cm over the suprapubic area before treatment. B, Complete resolution of the plaque with residual postinflammatory hyperpigmentation after 6 months of treatment.
Results of routine hematological and biochemical investigations and chest radiography were normal. A Mantoux test showed an induration of 2 × 1 mm at 48 hours. Direct potassium hydroxide preparation of the skin tissue showed multiple budding yeast cells and arthrospores (Figure 2A). Skin biopsy showed a granulomatous infiltrate composed of giant cells, lymphocytes, and histiocytes admixed with neutrophils and eosinophils. There were some hyaline hyphae branching at acute angles with multiple spores and budding yeasts. These structures stained with silver methenamine and periodic acid–Schiff stains (Figure 2B). Cultured skin biopsy specimens on 2 different occasions grew Trichosporon species identified by standard mycology laboratory procedures, including morphologic characteristics identified on cornmeal Tween-80 agar (Thermo Fisher Scientific Inc), color reduction on triphenyltetrazolium chloride medium, hydrolysis of urea, and sugar assimilation tests (Figure 2C). Using the molecular techniques of polymerase chain reaction analysis and DNA sequencing of internal transcribed spacer and intergenic spacer regions, the fungus species was identified as Trichosporon mycotoxinivorans.
A, Potassium hydroxide preparation of the skin tissue specimen showing multiple budding yeast cells and hyaline hyphae (original magnification ×400). B, Periodic acid–Schiff staining shows mixed granulomatous inflammatory infiltrate of giant cells, lymphocytes, and histiocytes admixed with neutrophils and eosinophils along with fungal spores and septate hyphae (original magnification ×200). C, Culture plate showing dried Trichosporon colonies.
Since there was no clinical and laboratory evidence of involvement of other organ systems, we made a diagnosis of primary cutaneous trichosporonosis. We treated the patient with itraconazole, 200 mg, twice daily, which was followed by moderate healing on the hand and face. However, after 5 months, she developed a skin-colored, soft, 8 × 12-cm swelling over the left elbow. Radiography showed no bony involvement. Fine-needle aspiration biopsy from the swelling revealed septate hyphae with some spores suggestive of a trichosporonotic abscess.
Drug susceptibility results showed that the fungus was sensitive to voriconazole but resistant to other antifungal drugs. Minimum inhibitory concentrations for fluconazole, caspofungin, and flucytosine were greater than 64 μg/mL, and for itraconazole, amphotericin, and micafungin, they were greater than 32µg/mL. Itraconazole treatment was discontinued, and voriconazole, 200 mg, twice daily was administered. Over the next 6 months, the patient had almost complete clearance of all her lesions with postinflammatory pigmentation and significant improvement in quality of life (Figure 1B). Voriconazole treatment was discontinued after 6 months, and at last follow-up 3 months later, there had been no recurrence.
Trichosporonosis most commonly presents as white piedra, a superficial infection of the hair shaft. Invasive trichosporonosis is known to develop in immunosuppressed patients and presents as a severe systemic illness. The least common manifestation is primary invasive cutaneous infections by Trichosporon species.1- 5
Trichosporon mycotoxinivorans was commonly used in various textile, agriculture, cosmetic, and pharmaceutical industries for its ability to detoxify mycotoxins such as ochratoxin A and zearalenone. In recent years, it has been isolated as a respiratory pathogen causing pneumonia in some cases of cystic fibrosis, but to our knowledge, a cutaneous manifestation has not been previously described.6 We could not establish a route of exposure in our patient.
Azoles have proven to be the most effective treatment options for other Trichosporon infections.1- 5 Our patient showed significant improvement of skin lesions with voriconazole.
Corresponding Author: M. Ramam, MD, Department of Dermatology, All India Institute of Medical Sciences, New Delhi, India (firstname.lastname@example.org).
Published Online: June 10, 2015. doi:10.1001/jamadermatol.2015.1354.
Conflict of Interest Disclosures: None reported.
Bhari N, Xess I, Pandey M, Arava S, Ramam M. Primary Cutaneous Trichosporonosis Responsive to Voriconazole. JAMA Dermatol. 2015;151(10):1139-1141. doi:10.1001/jamadermatol.2015.1354