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Mycopathologia 158: 53–56, 2004. 53 2004 Kluwer Academic Publishers. Printed in the Netherlands.

Sporotrichosis successfully treated with terbinafine and : Case report and review of the literature

Basak Coskun1, Yunus Saral1, Nusret Akpolat2, Arzu Ataseven1 & Demet C¸ic¸ek1 1Department of Dermatology, Faculty of Medicine, Frat University, Elazig, Turkey; 2Department of Pathology, Faculty of Medicine, Frat University, Elazig, Turkey

Received 21 October 2003; accepted in revised form 11 March 2004

Abstract is rare in Turkey. We report a 40-year-old woman who had subcutaneous sporotrichosis caused by schenckii that was successfully treated with terbinafine (250 mg, twice a day) for a period of 6 months. She received a saturated solution of orally for two months. Terbi- nafine and potassium iodide are suggested to be the agents of choice for treatment of subcutaneous sporotrichosis.

Key words: , terbinafine, potassium iodide

Introduction ulcers of different shapes and sizes on right arm and axilla; the largest one was about 4 cm in size Lymphocutaneous sporotrichosis is a chronic (Figure 1). She also had three suppurative nod- infection that results from accidental skin inocu- ules, 4 cm in size on right arm (Figure 2). The le- lation with the sporothrix schenckii, which sions had started as small nodules and perforated is often present on wood splinters or thorns [1]. with a purulent discharge and the recovered, but The lesions usually begin as a papule, pustule or reappeared as a similar lesion in another region of nodule, which may subsequently ulcerate. The her arm. She did not recall a definite history of organisms spread linearly through the lymphatic preceding trauma at the site of the lesion. She had system to regional lymph nodes. The resulting le- otherwise been in good health. There was no sions are usually painless subcutaneous nodules. history of taking immunosuppressive agents, risk S. schenckii is a , -like in factors for human immunodeficiency virus infec- tissue and filamentous in culture [1, 2]. We report a tion or diabetes mellitus. Informed consent of the 40-year-old woman who had subcutaneous spo- patient was obtained. This study was approved by rotrichosis caused by sporothrix schenckii, which the local ethics committee. is a rare occurrence in Turkey, and suggest an Laboratory investigations showed no abnor- approach for therapy. malities in full blood count, erythrocyte sedimen- tation rate, blood chemistry, urine analysis, and a skin test with tuberculin PPD was negative. Case report The punch biopsy specimens were taken from the edge and the centre of the largest lesion. The A 40-year-old woman was referred to our outpa- former was submitted for histopathological ex- tient department with a year history of papulon- amination and the latter used for bacterial culture. odular, erythematous lesions ranging in size from They were cultured in blood-chocolate-eou- 1 to 4 cm. On clinical examination, there were sin_methylen blue agars, and lowenstein-jensen 54

Figure 1. Ulcers of different shapes and sizes are seen on right Figure 3. Suppurative granulomatous inflammation (arrows) arm; the largest one was about 4 cm in size. (hematoxylin and eosin, ·40).

colonies showed some brown or black pigmenta- tion. Thus subcutaneous-type sporotrichosis was diagnosed. Additional immunological or other molecular methods were not used. The patient was treated with terbinafine (250 mg, twice a day) for a period of 6 months and she took a saturated solution of potassium iodide orally, at a dose of 10 drops three times daily and is increased as tolerated to 40–50 drops three times daily for 2 months. The response was rapid and the lesions healed after using potassium iodide.

Figure 2. Three suppurative nodules, 4 cm in size are found on right arm. Discussion agar was used for mycobacterial culture. The his- Sporotrichosis is a chronic infection of the cutane- topathology showed a marked suppurative gran- ous and subcutaneous tissues and lymphatics ulomatous inflammation (Figure 3). Fungal caused by the thermally dimorphic fungus, elements were not visible in serial sections stained S. schenckii. The organism grows saprophytically in with grocott-gomori methenamine silver. Direct nature on decaying vegetable matter and is found microscopy of biopsy specimens in potassium world-wide. The infection is initiated by traumatic hydroxide with lactophonel cotton blue and Gram inoculation of the organism into the skin. The dis- stain revealed yeast forms that were variable in ease is common among miners, forestry workers, shape, approximately 5–10 lm in diameter, with gardeners and florists. Host factors related to multiple buds. Gram and Ziehl–Neelsen stain of nutrition, , or sex may predispose certain smears from biopsy specimens excluded bacteria individuals to infection. The occurrence in a middle- and acid-fast pathogens, respectively. aged, white male who may have a history of alco- Bacterial and mycobacterial cultures showed holism was described as the alcoholic -gardener no growth. Fungal culture was positive. The fun- syndrome [3]. Our patient did not recall a definite gus grew readily on Sabouraud agar history of preceding trauma at the site of the lesion, (SDA) within 5 Days at 25 and 37 C (sporo- and there was no history of taking immunosup- trichosis thermal dimorphic fungi grow in mycelial presive agents, risk factors for human immunode- and yeast forms at 25 and 37 C, respectively). The ficiency virus infection or diabetes mellitus. colonies were initially cream-coloured, moist, Sporotrichosis has been reported from loca- yeast-like with a wrinkled surface; with age, the tions around the world, but most case reports 55 come from the tropical and subtropical regions of ole and two cases treated with potassium iodide, the Americas. The highest incidence in the world is and stated that these two treatments were similarly in Mexico. Most cases occur among the rural, effective; the former required 9–15 weeks and the peasant populations in the endemic areas of latter 10–14 weeks until healing [13]. Mexico, Central America and Brazil [4]. Sporo- Terbinafine 250 mg twice daily is an effective trichosis is frequently seen in the geographical re- treatment for cutaneous disease, with a rapid re- gions outlined above and rare in Turkey. Number sponse and high cure rates being reported by a of cases reported is therefore quite limited and number of authors [14, 15]. 400 mg protocols for treatment are not experienced. daily cured 71% of 14 patients with lymphocuta- However, sporotrichosis is rare in Turkey. A neous disease, but a smaller percentage of extra- search of online Turkish literature revealed only cutaneous cases, and is therefore considered only three report of an association of sporothrix sche- moderately effective in the treatment of sporotri- nckii with human disease [5]. chosis [16, 17]. Sporotrichosis may be confused with bacterial It is thought that KI + treatments pyoderma, inflammatory infections, give better results than anti-fungal treatment alone nocardiosis, leishmaniasis, , chro- [8]. It was thought that application of KI would moblastomycosis, and cutaneous tuberculosis [6]. enhance the efficacy of terbinafine treatment in the In our patient, the initial diagnosis was a lupus acute phase, and therefore, both were started at vulgaris, lichen planus, fungoides. the same time. This therapy was ceased when su- A saturated solution of potassium iodide has puration in the lesions of the patient regressed, but been used to treat cutaneous sporotrichosis since terbinafine was further continued for a period of the early 1900s, and it is still the mainstay of four months. Our patient treated with terbinafine treatment in most endemic areas. It is effective and (250 mg, twice a day) for a period of 6 months and inexpensive; however, the dosage regimen is com- she took a saturated solution of potassium iodide plicated, and common side-effects include metallic orally, at a dose of 10 drops three times daily and taste, enlargement and . Treat- is increased as tolerated to 40–50 drops three times ment is usually initiated with five drops three times daily for two months. The response was rapid and daily and is increased as tolerated to 40–50 drops the lesions healed after using potassium iodide. three times daily. Treatment should be continued In summary, sporotrichosis is rare in Turkey. for at least four weeks after apparent clinical We suggest that terbinafine and saturated solution ‘cure’. Some patients are allergic to , thus of potassium iodide should be the therapy of thermotherapy can be a useful alternative or ad- choice for cutaneous sporotrichosis. junct therapy [7–9]. 100–200 mg daily is the treatment of choice for cutaneous sporotrichosis, in situ- References ations where cost does not preclude its use. For fixed cutaneous or lymphocutaneous disease 1. Davis BA. Sporotrichosis. Dermatol Clin 1996; 14: 69–76. treatment should continue for 3–6 months, and 2. Kinbara T, Fukushiro R. 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