Disseminated Cutaneous Sporotrichosis with Fungal Sinusitis As An Initial Presentation of Underlying Myeloproliferative Neoplasm

Summary@#Sporotrichosis is a rare and chronic granulomatous subcutaneous mycotic infection caused by a dimorphic fungus, Sporothrix schenckii. We describe a patient with disseminated cutaneous sporotrichosis who was later diagnosed with myeloproliferative neoplasm and discuss the challenges and importance in diagnosing this rare condition.

Esporotricosis cutánea diseminada / Cutaneous disseminated sporotrichosis

La esporotricosis es una micosis profunda producida por especies del género Sporothrix, que presenta varias formas clínicas siendo la linfangítica la más frecuente. Las formas diseminadas son raras, están generalmente relacionadas a inmunosupresión y pueden ser solo cutáneas o sistémicas. Presentamos el caso de una mujer anciana que consulta por lesiones diseminadas en la piel e incluso afectación conjuntival producidas por Sporothrix globosa, con varios factores de inmunosupresión.

Sporotrichosis is a deep mycosis produced by species of the genus Sporothrix, that present several clinical forms, with lymphangitis being the most frequent. Disseminated forms are rare, are also related to immunosuppression and may be only cutaneous or systemic. We present the case of an elderly woman who consults for disseminated skin lesions and even conjunctival involvement caused by Sporothrix globosa, with several immunosuppression factors.

Esporotricosis diseminada, con compromiso cutáneo y visceral / Disseminated sporotrichosis, with cutaneous and visceral involvement

Se presenta un caso de esporotricosis cutánea diseminada, con compromiso visceral, en un hombre de 38 años, sin antecedentes de alteraciones previas de la inmunidad. Las lesiones cutáneas fueron nódulos y gomas que evolucionaron a úlceras y se acompañaron de adelgazamiento, fiebre, anemia, edemas, hepatosplenomegalia, adenomegalias múltiples, infiltrados pulmonares excavados, hipoalbuminemia, hematuria y albuminuria. El diagnóstico fue confirmado por el aislamiento deSporothrix schenckii de la secreción de los gomas y la detección de anticuerpos específicos en las pruebas de inmunodifusión en el gel de agar y contrainmunoelectroforesis con inmunodifusión secundaria en agarosa frente a una esporotriquina metabólica. La cepa aislada fue identificada comoSporothrix schenckii sensu stricto. Fue tratado con anfotericina B desoxicolato, anfotericina B en dispersión coloidal e itraconazol por vía oral con buena respuesta clínica.

A case of cutaneous disseminated sporotrichosis with visceral involvement is presented. The patient was a 38 year old man, without previous immunologicalalterations. He presented several skin nodules and gummata which evolved to skin ulcers. He also exhibited fever, weight loss, anemia, edema, hepatosplenomegaly, and multiple adenomegalies, excavated infiltrates in the right lung, hypoalbuminemia,albuminuria and hematuria. Sporothrix schenckii was isolated from gummatasecretion of several skin lesions and specific antibodies were detected byimmunodiffusion in agar gel and counterimmunoelectrophoresis with secondary immunodiffusion in agarose, using a S. schenckii metabolic antigen. The isolate was identified as Sporothrix schenckii sensu stricto, which is the most frequently isolated in Argentina. The patient was treated with amphotericin B-deoxycholate and amphotericin B colloidal dispersion intravenously, later he received itraconazole by oral route with good clinical response.

A Case of Cutaneous Disseminated Type of Sporotrichosis Resistant to Itraconazole and Terbinafine / 대한의진균학회지

Sporotrichosis, a deep fungal infection caused by Sporothrix schenckii, is mainly treated with potassium iodide (Kl) and other antifungal agents, and recently there were reports of cases treated with itraconazole or terbinafine. A 56-year-old female patient presented with multiple painless firm nodules covered with crusts measured 1.0x1.5 cm and 1.0x3.0 cm on the right side of neck and an erythematous plaque with ulceration measured 2.0x2.0 cm on the right cheek for 20 days. Eight months before visit, she noted two subcutaneous nodules on the right side of neck. Excisional biopsy and treatment for tuberculous lymphadenitis were done. There had been no clinical response for 2 months, but new painless nodules and plaque with ulceration were developed on the opposite side of previous lesions. Biopsy showed a granulomatous change with polymorphic infiltrates, yeast cells and asteroid body. Routine laboratory findings and chest X-ray were not contributory. No evidence of tuberculosis was found in the examination with sputum and tissue.4 dark brown to black, moist and wrinkled colony on the Sabouraud's dextrose agar and septate, branched mycelia and clustered conidia on slide culture were shown to be consistent with Sporothrix schenckii. Cutaneous disseminated type of sporotricosis was diagnosed. The treatment was started with 500 mg of terbinafine daily which was known to be fungicidal, but there was no improvement clinically and mycologically for 2 months except initial transient stopping of progression. The regimen was subsequently changed to intraconazole 200 mg daily for 3 months, but new lesions were developed. So she was treated with saturated solution of potassium iodide. Five drops were given three times a day, and the dose was increased by 5 drops each day to a dose of 30 drops three times a day. Nodular lesions markedly decreased in size in two weeks, but serial tissue cultures were positive. After 3 months of administration, tissue culture was negative. No evidence of clinical relapse was noted after 3 years of follow-up.