MYCOSES
ACCEPTED: OCTOBER 24, 1991
35,43-45 (1992)
CASEREPORT
Symmetrical deforming cutaneous sporotrichosis of long duration
Symmetrische, deformierende, langdauernde Sporotrichose der Haut R. Hernandez Perez’, E. Borregales Torres2, M. Garcia de Mendez2, E. Sauerteig3 and K. Salfelder3 Key words. Sporothrix schenckii, sporotrichosis, long duration, histopathology. Schliisselworter. Sporothrix schenckii, Sporotrichose, lange Dauer, Histopathologie.
Summary. A symmetrical deforming cutaneous sporotrichosis with verrucous lesions on both hands and of long clinical duration is reported. The difficulties in making an etiological diagnosis are considered. Zusammedassung. Es wird der Fall einer symmetrischen Sporotrichose mit ausgedehnten, deformierenden, verrukosen Veranderungen an beiden Handen mit langem Verlauf beschrieben. Es wird besonders auf die Schwierigkeiten eingegangen, eine atiologische Diagnose zu stellen.
cutaneous manifestations with lymphangitic satellite lesions and sometimes verrucous skin alterations were seen. Single, localized skin nodules or ulcers were occasionally found. Involvement of mucosae and of extracutaneous sites was not observed in the cases of sporotrichosis we examined. The unusual and extensive skin lesions and the difficulties of clinical diagnosis were motives to report this case.
Case report Introduction
A 58-year-old agricultural worker who frequently
Sporotrichosis, a cosmopolitan infection endemic in the tropics and in developing countries, has recently been reported from some industrialized countries. The pleomorphic cells of Sporothrix schenckii cannot be detected in tissues in cases with a chronic course. Only occasionally do asteroid bodies, containing fungus cells, lead to histologic diagnosis. In Mirida and Barinas, Venezuela the classical
’
Departamento de Dermatologia, Hos ital Universitario Luis Bazetti, Barinas, Venezuela, and Departamento de Microbiologia, Secci6n Micologia, Facultad de Farmacia and 3Laboratorio de Investigaci6n en Patologia, Facultad de Medicina, Universidad de 10s Andes, Mtrida, Venezuela.
P
Correspondence: Dr Karl Salfelder, Apartado 68 1, Mtrida, Venezuela.
handled cattle and other animals (“matarife”, Spanish) lived in a hut in the wilderness of Barinas State, Venezuela in highly unhygienic conditions. H e developed lesions on the dorsal parts of both hands 8 years ago which soon became deforming (Fig. 1). He had to live alone, because he was rejected by family members and friends who could not support the aspect of his ugly hands . He was first examined in February 1991. In May 199 1, finally, etiologic diagnosis was achieved and treatment applied. Over the course of more than a year several attempts were made to culture clinical material and five biopsies were performed. Routine laboratory examinations, chest X-ray, electrocardiogram and intradermic tests (PPD and Montenegro) were negative. Clinically, diagnostic hypotheses were: chro-
44
R. HERNANDEZ P ~ R EETZ AL.
Mycology
Figure 1. Sporotrichosis: cutaneous verrucous lesions on both hands with marked deformation.
moblastomycosis, leishmaniasis, tuberculosis and atypical mycobacteriosis. In the first few months of 1991 the patient developed several nodules on the left arm and a crusted skin ulcer 2 cm in diameter on his abdomen.
Histology A marked pseudoepitheliomatous hyperplasia of epidermis was accompanied by dense and diffuse cellular infiltrates in dermis and subcutis with lymphocytes and granulocytes, plasma cells and mononuclear elements. Furthermore, microabscesses and a granulomatous reaction with epithelioid and giant cells were noted (Fig. 2). Haematoxylin and eosin stain and the methods of Ziehl-Neelsen, Gram, PAS and Grocott did not reveal micro-organisms. Asteroid bodies could not be detected. Several biopsies with numerous sections were examined and several pathologists in Venezuela and Germany could not make an etiologic diagnosis.
Figure 2 Sporotrichosis: typical “mixed” tissue reaction with microabscesses and granulomatous reaction. Fungi not visible. H&E stain.
Cultures of material from nails revealed Trichophyton rubrum. I n scrapings of skin, elements of Malassezia f u f u r were found. In several attempts to culture tissues from the verrucous lesions no pathogenic fungi were found, until in May 1991 in a last effort the patient was brought to MCrida and again cultures were done on Sabouraud glucose agar. Within a few days colonies of Sporothrix schenckii (Fig. 3) and Candida sp. were detected. This isolated strain of S. schenckii developed black pigment in a few days at room temperature (20°C). In order to confirm diagnosis of sporotrichosis a sporotrichin skin test was performed and culture material was inoculated into rats subcutaneously. Skin test and inoculation were strongly positive.
Treatment Potassium iodide was given orally, initially 0.5 day-’ with an increasing dosage after 2 weeks up to 5 g day-‘, for three months with remarkable improvement of the cutaneous lesions (Fig. 4). In August 1991 treatment with itraconazole was started to complete the curing process.
Discussion The unusual skin manifestations and a clinical course of long duration seen in our case are features which have been documented before [ 1-51. Lymphangiti.c spread and apparently dissemination by inoculation (scratching) occurred on the abdomen in our case only after some time. Infection may have taken place through contact with animals, since natural infection in animals is known and the patient had close contact
Figure 3. Sporotrichosis: mount of culture of Sporofhhrix schmkii at 20 “C with hyphae and conidia. Typical diagnostic “daisy” form. Lactophenol blue.
mycoses 35, 43-45 (1992)
CUTANEOUS SPOROTRICHOSIS
45
schenckii are, however, found easily in tissues in acute stages of infection and in cases of local immunosuppression [3, 61. Failure to grow the fungus in culture in the first months after the patient came to the clinic was apparently due to inexperience of the laboratory personnel.
References
Figure 4. Marked improvement of skin lesions after treatment for three months with potassium iodide.
with them. The symmetrical infection on both hands may have occurred simultaneously from the beginning. One of the principal motives of this publication was to emphasize again the lack of fungal cells in tissues in cases of a chronic course of this mycosis. Therefore histologic diagnosis of this type of sporotrichosis cannot be achieved, and cultures and inoculations into animals must be performed in order to produce a clinical diagnosis when histology is negative. Cells of S’orothrix
mycoses 35, 43-45 (1992)
Itoh, M., Okamot, S. & Kariya, H. (1966) Survey of 200 cases of sporotrichosis. Dermatologica 172, 209-2 13. Londero, A. T. & Ramos, C. D. (1980) Esporotricose: Estudo de 195 casos observados no interior do Rio Grande do Sul. R. AMRIGS (Porto Alegre) 24, 104-106. Salfelder, K., de Liscano, T. R. & Sauerteig, E. ( 1 990) Atlas of Fungal Pathology. Dordrecht: Kluwer Academic Publishers. Velasquez, J. P., Restrepo, A. & Calle, G. (1976) Experiencia de 12 aiios con la esporotricosis. Polimorfismo clinic0 de la entitad. Anlioquia MPdica 26, 153-169. Villaqa-Neto, C. M., Rossetti, R. B., Fishman Gompertz, 0. & Paschoal, L. H. C. (1988) Localized cutaneous verrucous sporotrichosis of 26 years duration. mycoses 31, 353-355. Mohri, S. (1987) Study in sporotrichosis. I. Statistical study in sporotrichosis-review of reports published from 1984 to 1985 and 100 cases from our clinic. Yokohama Med. Bull. 38, 19-26.
ACCEPTED: OCTOBER 24, 1991
35,43-45 (1992)
CASEREPORT
Symmetrical deforming cutaneous sporotrichosis of long duration
Symmetrische, deformierende, langdauernde Sporotrichose der Haut R. Hernandez Perez’, E. Borregales Torres2, M. Garcia de Mendez2, E. Sauerteig3 and K. Salfelder3 Key words. Sporothrix schenckii, sporotrichosis, long duration, histopathology. Schliisselworter. Sporothrix schenckii, Sporotrichose, lange Dauer, Histopathologie.
Summary. A symmetrical deforming cutaneous sporotrichosis with verrucous lesions on both hands and of long clinical duration is reported. The difficulties in making an etiological diagnosis are considered. Zusammedassung. Es wird der Fall einer symmetrischen Sporotrichose mit ausgedehnten, deformierenden, verrukosen Veranderungen an beiden Handen mit langem Verlauf beschrieben. Es wird besonders auf die Schwierigkeiten eingegangen, eine atiologische Diagnose zu stellen.
cutaneous manifestations with lymphangitic satellite lesions and sometimes verrucous skin alterations were seen. Single, localized skin nodules or ulcers were occasionally found. Involvement of mucosae and of extracutaneous sites was not observed in the cases of sporotrichosis we examined. The unusual and extensive skin lesions and the difficulties of clinical diagnosis were motives to report this case.
Case report Introduction
A 58-year-old agricultural worker who frequently
Sporotrichosis, a cosmopolitan infection endemic in the tropics and in developing countries, has recently been reported from some industrialized countries. The pleomorphic cells of Sporothrix schenckii cannot be detected in tissues in cases with a chronic course. Only occasionally do asteroid bodies, containing fungus cells, lead to histologic diagnosis. In Mirida and Barinas, Venezuela the classical
’
Departamento de Dermatologia, Hos ital Universitario Luis Bazetti, Barinas, Venezuela, and Departamento de Microbiologia, Secci6n Micologia, Facultad de Farmacia and 3Laboratorio de Investigaci6n en Patologia, Facultad de Medicina, Universidad de 10s Andes, Mtrida, Venezuela.
P
Correspondence: Dr Karl Salfelder, Apartado 68 1, Mtrida, Venezuela.
handled cattle and other animals (“matarife”, Spanish) lived in a hut in the wilderness of Barinas State, Venezuela in highly unhygienic conditions. H e developed lesions on the dorsal parts of both hands 8 years ago which soon became deforming (Fig. 1). He had to live alone, because he was rejected by family members and friends who could not support the aspect of his ugly hands . He was first examined in February 1991. In May 199 1, finally, etiologic diagnosis was achieved and treatment applied. Over the course of more than a year several attempts were made to culture clinical material and five biopsies were performed. Routine laboratory examinations, chest X-ray, electrocardiogram and intradermic tests (PPD and Montenegro) were negative. Clinically, diagnostic hypotheses were: chro-
44
R. HERNANDEZ P ~ R EETZ AL.
Mycology
Figure 1. Sporotrichosis: cutaneous verrucous lesions on both hands with marked deformation.
moblastomycosis, leishmaniasis, tuberculosis and atypical mycobacteriosis. In the first few months of 1991 the patient developed several nodules on the left arm and a crusted skin ulcer 2 cm in diameter on his abdomen.
Histology A marked pseudoepitheliomatous hyperplasia of epidermis was accompanied by dense and diffuse cellular infiltrates in dermis and subcutis with lymphocytes and granulocytes, plasma cells and mononuclear elements. Furthermore, microabscesses and a granulomatous reaction with epithelioid and giant cells were noted (Fig. 2). Haematoxylin and eosin stain and the methods of Ziehl-Neelsen, Gram, PAS and Grocott did not reveal micro-organisms. Asteroid bodies could not be detected. Several biopsies with numerous sections were examined and several pathologists in Venezuela and Germany could not make an etiologic diagnosis.
Figure 2 Sporotrichosis: typical “mixed” tissue reaction with microabscesses and granulomatous reaction. Fungi not visible. H&E stain.
Cultures of material from nails revealed Trichophyton rubrum. I n scrapings of skin, elements of Malassezia f u f u r were found. In several attempts to culture tissues from the verrucous lesions no pathogenic fungi were found, until in May 1991 in a last effort the patient was brought to MCrida and again cultures were done on Sabouraud glucose agar. Within a few days colonies of Sporothrix schenckii (Fig. 3) and Candida sp. were detected. This isolated strain of S. schenckii developed black pigment in a few days at room temperature (20°C). In order to confirm diagnosis of sporotrichosis a sporotrichin skin test was performed and culture material was inoculated into rats subcutaneously. Skin test and inoculation were strongly positive.
Treatment Potassium iodide was given orally, initially 0.5 day-’ with an increasing dosage after 2 weeks up to 5 g day-‘, for three months with remarkable improvement of the cutaneous lesions (Fig. 4). In August 1991 treatment with itraconazole was started to complete the curing process.
Discussion The unusual skin manifestations and a clinical course of long duration seen in our case are features which have been documented before [ 1-51. Lymphangiti.c spread and apparently dissemination by inoculation (scratching) occurred on the abdomen in our case only after some time. Infection may have taken place through contact with animals, since natural infection in animals is known and the patient had close contact
Figure 3. Sporotrichosis: mount of culture of Sporofhhrix schmkii at 20 “C with hyphae and conidia. Typical diagnostic “daisy” form. Lactophenol blue.
mycoses 35, 43-45 (1992)
CUTANEOUS SPOROTRICHOSIS
45
schenckii are, however, found easily in tissues in acute stages of infection and in cases of local immunosuppression [3, 61. Failure to grow the fungus in culture in the first months after the patient came to the clinic was apparently due to inexperience of the laboratory personnel.
References
Figure 4. Marked improvement of skin lesions after treatment for three months with potassium iodide.
with them. The symmetrical infection on both hands may have occurred simultaneously from the beginning. One of the principal motives of this publication was to emphasize again the lack of fungal cells in tissues in cases of a chronic course of this mycosis. Therefore histologic diagnosis of this type of sporotrichosis cannot be achieved, and cultures and inoculations into animals must be performed in order to produce a clinical diagnosis when histology is negative. Cells of S’orothrix
mycoses 35, 43-45 (1992)
Itoh, M., Okamot, S. & Kariya, H. (1966) Survey of 200 cases of sporotrichosis. Dermatologica 172, 209-2 13. Londero, A. T. & Ramos, C. D. (1980) Esporotricose: Estudo de 195 casos observados no interior do Rio Grande do Sul. R. AMRIGS (Porto Alegre) 24, 104-106. Salfelder, K., de Liscano, T. R. & Sauerteig, E. ( 1 990) Atlas of Fungal Pathology. Dordrecht: Kluwer Academic Publishers. Velasquez, J. P., Restrepo, A. & Calle, G. (1976) Experiencia de 12 aiios con la esporotricosis. Polimorfismo clinic0 de la entitad. Anlioquia MPdica 26, 153-169. Villaqa-Neto, C. M., Rossetti, R. B., Fishman Gompertz, 0. & Paschoal, L. H. C. (1988) Localized cutaneous verrucous sporotrichosis of 26 years duration. mycoses 31, 353-355. Mohri, S. (1987) Study in sporotrichosis. I. Statistical study in sporotrichosis-review of reports published from 1984 to 1985 and 100 cases from our clinic. Yokohama Med. Bull. 38, 19-26.
