Nodular granulomatous phlebitis of the penis in a patient with antecedent tuberculous epididymitis: The first reported case

Abstract

pustule. The incisional biopsy was performed on the dorsum of the left hand and, histologically, mixed inflammatory infiltrates and cyst-like structures were visible in the dermis. Fungal hyphae were positive by Grocott–Gomori methenamine silver and periodic acid–Schiff stains (Fig. 1b,c). She was treated with oral itraconazole 200 mg/day and i.v. cefbuperazone 2 g/ day, and the pustules were punctured and drained. After 4 weeks, cottony fungal colonies were cultured and the specific species was identified as Pseudallescheria angusta by using sequence analysis of the internal transcribed spacer gene. We checked thoracoabdominal computed topography to evaluate solid organ involvement including the lungs, which are the most common site of infection, and found no abnormality. Although pustules and papules of the lesion were slightly improved by oral itraconazole treatment, it was still accompanied by oozing and edema. We changed oral itraconazole to oral voriconazole 400 mg/day for 3 months, and there was no evidence of recurrence after 3 months of follow up (Fig. 1d,e). Scedosporium species usually cause infections in immunocompromised patients. The frequently involved sites are respiratory tract, eyes, central nervous system, skin and bone. The most common sites of cutaneous infection are the extremities, because these are usually caused after trauma. Skin involvement may be also an initial manifestation of disseminated infection. The treatment of choice of scedosporial infection is voriconazole monotherapy. The recommended i.v. regimen in adults is 6 mg/kg every 12 h for the first 24 h on day 1 followed by 4 mg/kg every 12 h; and the recommended oral regimen is 200 mg every 12 h. The recommended therapy duration varies depending on host immunity, site and extent of infection. Isavuconazole and posaconazole have some in vitro activity but amphotericin B is usually resistant to Scedosporium. Surgical debridement of the necrotic tissue and drainage of the purulent material can be helpful. There was a case of P. angusta infection in pulmonary mycetoma from a patient with fibrocystic sarcoidosis and he was treated by intracavitary voriconazole. In the present case, the lesions were improved by itraconazole treatment but there was still oozing and edema. Thus, we changed itraconazole to oral voriconazole which is generally considered effective. After 2 months of treatment with voriconazole, there was no evidence of recurrence on the left hand after 2 months of follow up. Scedosporial infections are often reported in immunocompromised patients, but they are uncommon in immunocompetent patients. We report the first case of cutaneous P. angusta infection successfully treated with voriconazole in an immunocompetent patient.