Lymphocutaneous Sporotrichosis in an Immunocompetent Patient: A Case Report from Extreme Southern Italy
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- Criseo, G., Malara, G., Romeo, O. et al. Mycopathologia (2008) 166: 159. doi:10.1007/s11046-008-9121-4
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Sporotrichosis is a mycotic infection rarely encountered in European countries, including Italy. We describe lymphocutaneous sporotrichosis in an immunocompetent 64-year-old male Italian mason. The patient had linear nodules along the right upper arm with severe lymphadenopathy in the same arm. Sporothrix schenckii was identified by phenotypic and molecular methods. Treatment with itraconazole and methylprednisolone followed by administration of itraconazole alone successfully cured the infection. To our knowledge, this is the first documented report of sporotrichosis from Calabria and Sicily, extreme southern Italy.
KeywordsLymphocutaneous SporotrichosisSporothrix schenckiiSporotrichosis in ItalyItraconazole
Sporothrix schenckii is a dimorphic fungus that occurs in mold form in the natural environment (∼24°C), but it grows as a yeast-like form in infected tissues or in culture at 37°C. The fungus can be found as a saprobe in dead or senescent vegetation in soil (e.g. thorns, straw, wood, dry leaves) , in water, or in several animals, including equines, pigs, birds, cats, and dogs [2–4].
Sporotrichosis, a mycotic infection caused by S. schenckii, is frequently found in outdoor workers (e.g. farmers, florists, gardeners, and miners) . This disease, described for the first time by Schenck in 1898, is also known as Schenck’s disease or even Rose Gardener’s Disease. This infection occurs in many countries throughout the world, but it is more common in tropical and subtropical places with high humidity such as North and South America , South Africa , and Australia ; however, it is rare in Europe [1, 9].
In Italy, 16 cases of sporotrichosis have been reported in the period 1978–1992 in the province of Bari, Apulia, Southern Italy , whereas only few cases have been described in other Italian regions for many years now [11, 12].
Most of the time the infection is cutaneous or subcutaneous as a consequence of a traumatic implantation of S. schenkii onto the skin; less frequently it occurs as an osteoarthritis or disseminated form.
We describe lymphocutaneous sporotrichosis in an Italian immunocompetent patient who had linear nodules along the right upper arm with severe lymphadenopathy in the same arm.
The pustula rapidly developed into several painful nodules along the right upper extremity of the hand and near the elbow in the upper right arm (Fig. 1a). Cutaneous examination showed several tender linear erythematous nodule-ulcerative lesions that started from the little finger and reaching the shoulder of the same side. There were regional edema and lymphadenopathy and sometimes short periods of fever (temperature: 38–39°C). Routine laboratory investigations, including Mantoux test, were normal, with the exception of the white cells (15.2 × 103/μl), ESR (1° h:30 mm), and C-reactive protein (3.17 mg/dl) that were slightly high. Neither concomitant diseases nor clinical signs of immunosuppression were observed in this patient.
At this point sporotrichosis was suspected by one of us. A mycological study to isolate Sporothrix schenckii was carried out according to the classical procedure described by Travassos and Lloyd . Briefly, specimens of pus and wound swabs were plated in duplicate onto Sabouraud 2% dextrose agar (SDA), SDA supplemented with chloramphenicol (0.1%), and SDA with chloramphenicol (0.1%) and cycloheximide (0.1%). The cultures were incubated at 28°C and 37°C for 5–7 days, respectively.
After 96 h, culture dishes incubated at 28°C developed several cream-colored, smooth or verrucous, moist colonies with irregular borders and traces of a darker pigmentation and occasional aerial mycelia. Then, the colonies became dark as shown in Fig. 2c,d.
Cultures incubated at 37°C yielded small yeast cream-colored colonies. Direct microscopic examination of fungal cultures revealed abundant delicate and intermeshed mycelial hyphae; there were piriform spores adhering to sterigmata and producing a cuff-like sheath round a hypha, at the tip of which they formed a rosette (Fig 2a). In the yeast-like colonies the cells were round, oval, or spindle- or cigar-shaped and produced one or more budding cells. On the basis of these morphological and cultural features we identified the strains as S. schenckii.
Additional molecular confirmative test, using PCR amplification of the chitin synthase 1 gene , confirmed the identity of the fungus.
In vitro susceptibility to itraconazole and amphotericin B was performed by broth microdilution test according to Kohler et al. . The MICs of itraconazole and amphotericin B were of 4 μg/ml and 8 μg/ml, respectively.
We decided to initiate treatment with oral itraconazole (200 mg daily). Since lymphadenopathy did not go away, 20 mg daily of i.m. methylprednisolone (Urbason) was also administered to obtain a lowering of local tissue edema. After six weeks of treatment we have seen a good improvement of the patient’s condition; in fact the nodular lesions on the right arm had almost disappeared together with the lymphadenopathy. But a few weeks later the lymphadenopathy came back again and so we decided to increase the methylprednisolone dosage from 20 to 40 mg daily for one week while keeping the itraconazole at the same dosage. Finally, after another five weeks of the itraconazole treatment alone the patient was successfully cured (Fig. 1c).
Isolates of S. schenckii are rarely encountered in the clinical laboratory unless they are associated with sporotrichosis.
Sporotrichosis is a worldwide mycotic infection that occurs in temperate, tropical, and subtropical countries. It is rarely encountered in European countries, including Italy. However, in 2004 a small number of cases of sporotrichosis had been reported from Spain .
The relatively low incidence of diagnosed sporotrichosis in Europe could be simply because only few clinical laboratories perform routinely mycological isolation and/or identification of S. schenckii from skin samples. Moreover few studies focusing on the diffusion of S. schenckii in European countries were carried out.
Until 1992, a few cases of sporotrichosis have been described in Italy; most of them were concentrated in Apulia . Since then, only one case of cutaneous sporotrichosis was reported in Naples, Italy .
In our study we report a new case of sporotrichosis in southern Italy in a patient who did not show other concomitant diseases or immunosuppression.
Successful treatment with itraconazole of the present case point out that this antimycotic agent can be very effective, especially against lymphocutaneous sporotrichosis. This result agrees with those recently reported by several other authors [12, 17–19], including those following Infectious Diseases Society of America (IDSA) guidelines . In the present clinical case, although complete sporotrichosis healing was obtained by itraconazole treatment, concomitant corticoid administration has proved to be useful during the course of the lymphadenopathy even if itraconazole plus methylprednisolone is not a treatment of choice for sporotrichosis .
To our knowledge, this is the first documented report of lymphocutaneous sporotrichosis from Calabria and Sicily, extreme southern Italy. The occurrence of sporotrichosis in Italy, almost exclusively in the south of this country, provides evidence of the important role played by environmental factors in this area. This observation would seem to justify further researches into environment of this particular geographic area that could represent an ideal habitat for S. schenckii.