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Rev. Inst. Med. trop. S. Paulo vol. 39 no. 4 São Paulo July/Aug. 1997
Report of two cases and review of the literature
Luiz Carlos SEVERO, Flávio de Mattos OLIVEIRA & Alberto Thomaz LONDERO
Two cases of subcutaneous scedosporiosis, caused by Scedosporium apiospermum, are reported. Both patients had lesions localized in the forearm: a solitary ulceration in one and a sporotrichoid-like lesion in the other. The literature is reviewed.
KEYWORDS: Scedosporium apiospermum; Subcutaneous scedosporiosis; Subcutaneous mycosis.
Scedosporium apiospermum (teleomorph, Pseudallescheria boydii) is a largely widespread water and soil inhabiting fungus9 that causes a wide spectrum of diseases5. It may be classified among the pathogenic as well as among the opportunistic agents of mycoses21. It may also be a colonizer of the respiratory tract25 and, in addition, has been implicated in allergic bronchopulmonary conditions15.
Usually S. apiospermum proceeds as a pathogenic fungus when introduced traumatically into the skin or subcutaneous tissue of a normal host, causing mycetoma. But it may also elicit other types of subcutaneous lesion. However, in the immunosuppressed host, polymorphic cutaneous or subcutaneous lesions may result from the inoculation of the fungus.
Two cases of subcutaneous lesions caused by S. apiospermum will be reported and a review on the subject will be presented.
Case 1. A 73 year-old white woman was seen by an ulceration in his left forearm, in December 1986. She complained of dyspnea, weight loss (25 kg in 10 months), anorexia and weakness. Her medical history included a carcinoma of the breast (February 1986) treated with chemotherapy and prednisone (60 mg/day) and a pleural effusion at the right lung (March 1986), which was shown to be neoplastic by a thoracocentesis.
The ulceration on the flexor surface of the left forearm measured 1 × 2 cm, was limited by a smooth erythematous border and was covered by a yellowish crust (Fig. 1). A biopsy was performed. A potassium hydroxide preparation of a portion of biopsied tissue demonstrated hyaline, septate, dichotomously branched hyphae. Another part of the biopsy was planted onto Sabourauds dextrose agar incubated at room temperature. White floccose colonies were obtained. Microscopically solitary one-celled, pale brown ovoid conidia (4 to 9 × 6 to 12 µm) with truncate base on the apex of conidiophores (Scedosporium stage) were observed in slide culture.
Patients pulmonary function continued to deteriorate and she died before receiving antifungal therapy.
Fig. 1 - Patient 1. Forearm lesion showing ulceration with seropurulent discharge.
Case 2. A 66 year-old white woman presented with three painful subcutaneous nodules on the flexor surface of the left forearm, three months ago. The lesions became ulcerated and were covered by dark necrotic crusts. A biopsy was performed and a diagnosis of pyoderma gangrenosum was established, in December, 1994. In spite of antibioticotherapy and topical use of mupirocin, the lesions progressed to necrotic plaques (Fig. 2).
In March, 1995 another biopsy was performed. A chronic inflammatory reaction was observed. A potassium hydroxide preparation of the exudate revealed hyaline, branched, septate hyphae and conidia. These fungal elements were well stained by Grocott (Fig. 3). Biopsied tissue was also planted onto Sabouraud medium with chloramphenicol. White floccose colonies was obtained; microscopically the conidial state of Scedosporium were observed.
The patient was treated with itraconazole (100 mg/day); in six months the lesions healed, and treatment was continued for one year.
Fig. 2 - Hyperpigmented plaque lesion in forearm of patient 2.
Fig. 3 - Branching, septate hyphae and conidium (insert) seen in biopsied tissue smear (Case 2). GMS (x 1000).
Eleven cases of subcutaneous lesions caused by S. apiospermum could be gathered in the literature, two from Brazil8, 11. Patients data, localization and type of lesions, underlying conditions, diagnosis and treatment, including our cases, were shown in table 1.
Interesting findings on these 11 cases were as follows. Trauma at the site of the lesion and isolation of the fungus from the soil of the patients garden was quoted by Morin et al. (1987). Introduction of the fungus by surgical wounds was referred in three cases10,25. Corticotherapy was the underlying conditions in five patients4,10,11,17,26, hyperfunction of adrenal glands and diabetes in one patient3, and diabetes alone in another25. In nine patients the disease seems to be limited to the soft tissue; in one patient localized signs of arthritis and osteitis was disclosed by 99 mTc scan26; in the remaining patient, after being limited during 3 months, hematogenic disseminated lesions appeared in the opposite member and death occurred by respiratory failure in spite of no evidence of lung lesions17. Two patients presented lesions resembling those of lymphangitic sporothricosis4,26. It is interesting to refer that mycetoma can also, exceptionally, manifests as a sporotrichoid-like lesion18. Like our case nº 2, the patient reported by Van Hecke et al. had a histopathological diagnosis of pyoderma grangrenosum in the examination of the first biopsy.
Two other reported cases have been reported in the literature as subcutaneous S. apiospermum infection. However, in one of them27 the authors called into question if the cutaneous microabscess was not really the result of hematogenic dissemination of a non-elucidated pneumopathy. The other report29, based only on medico-legal protocols, deals with a patient with a two year (1917-1919) non-elucidated suppurative lesion on a finger; two years later (1921) a furuncular-like lesions appeared on the hand and forearm, associated with subcutaneous and muscular gumma-like lesions, arthritis, periostitis of many bones and orchiepidimitis; S. apiospermum was then isolated from an abscess; treated with vaccine obtained from the culture of the fungus the patients lesions healed; death occurred two years later (1923), after a septicemic-like disease.
On the other hand, cutaneous or subcutaneous lesions resulting from hematogenic dissemination S. apiospermum infections have also been reported. They manifest as erytemathous papular rash7, an eritemathous papular lesion with a necrotic center6, a solitary pustule24, or nodules with dusky, light green center1. Tumefaction24 or induration of the skin1, at the site of a catheter, has also been implicated as portal of entry of the fungus. Biopsy, direct examinations of pus and culture of these lesions led to the diagnosis of the mycosis.
Conidia of Scedosporium type are produced in the host tissue as well as in culture9. For that reason diagnosis of S. apiospermum or S. inflatum can be presumed on the basis of microscopic examination of clinical specimens in which hyphae and conidia are seen, as in our case nº 22,7,14,19,20,22,23,28. However, species identification of the agent requires the isolation of the fungus.
|Case (ref.)||Age, sex||Subcutaneous lesions||Underline condition||Diagnosis||Treatment; outcome|
|1 (12)||76, M||Soft tissue painful, erythematous swelling of the 4th and 5th finger; in one year, involved the hand, wrist and forearm. X-ray did not reveal bone lesions.||None||Culture of synovia and synovium||Amphotericin B and miconazole; relapse|
|2 (26)||76, M||Nodules (1 mm to 4 cm) from the dorsum of the 5th finger along the ulnar margin of the right forearm; arthritis and osteitis of the wrist, disclose by X-ray and 99mTc bone scan.||Prednisone, bronchitis, hypertension, temporal arteritis||Pus (direct, smears PAS); biopsy (Grocott) Culture of the pus||Ketoconazole; death: heart failure|
|3 (3)||59, F||Non tender, swollen, erythematous induration, covered by many pustular lesions on the righ leg 2 months duration.||Diabetes Cushings disease||Biopsy (PAS). Culture of the pus and biopsied fragments||Miconazole. Control of immunosuppression|
|4 (25)||46, M||Infected surgical wound resulting from partial hemilaminectomy.||None||Culture of infected tissue||Surgical debridement|
|5 (25)||64, M||Infected axillo femoral graft.||Diabetes||Culture of the graft||Graft resection|
|6 (10)||24, M||Purulent material draining from a craniotomy wound.||Dexamethazone||Culture of the pus||Miconazole|
|7 (16)||45, F||Ulceration (2 × 5 cm) covered by adherent slough on the dorsum of right ankle.||None||Direct and culture of slough biopsy||Surgical debridement and skin graft|
|8 (17)||64, M||Painless tumefaction and ulceration (1 cm) with erythematous border, covered by a necrotic scar and draining a yellowish pus, 3 month duration.||Waldenstroms disease Corticotherapy Cytotoxic drugs||Pus (direct), biopsy (Grocott), culture||Ketoconazole|
|9 (8)||38, M||Tumefaction of the right malleolar region with a fistula draining purulent material.||None||Culture of the pus||Surgical extraction of a rubber fragment|
|10 (11)||45, M||One painless, tender, flutuant nodule (2 cm) in the left knee 4 month duration.||Diabetes, renal transplantation||Biopsy (Grocott and culture) and corticotherapy||Surgery Itraconazole|
|11 (4)||73, M||Subcutaneous nodules distributed on the foot, tibial crest and tigh (0.5 to 3 cm), 4 month duration.||Hortons disease Corticotherapy||Biopsy (Grocott) Culture of the pus||Itraconazole|
|Case 1||73, F||Ulceration (1 × 2 cm) on the flexor surface of the forearm.||Carcinoma of the breast |
|Direct and culture||None; death: pulmonary failure|
|Case 2||66, F||Three painfull nodules on the flexor surface of the left forearm.||None||Direct, smears (Grocott) and culture||Itraconazole; cured.|
Scedosporiose subcutânea. Relato de dois casos e revisão da literatura
São relatados dois casos de scedosporiose subcutânea por S. apiospermum. Em ambos os pacientes as lesões localizavam-se no antebraço: em um deles, uma ulceração, no outro, uma lesão" esporotricóide". Foi feita revisão comentada da literatura pertinente.
1. BERNSTEIN, E.F.; SCHUSTER, M.G.; STIERITZ, D.D. et al. Disseminated cutaneous Pseudalescheria boydii. Brit. J. Derm., 132:456-460, 1995. [ Links ]
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3. COLLIGNON, P.J.; MacLEOD, C. & PACKHAM, D.R. Miconazole therapy in Pseudalescheria boydii infection. Aust. J. Derm., 26:129-132, 1985. [ Links ]
4. CREMER, G.; BOURNERIAS, I.; MHALLA, S. et al. Scédosporiose cutanée non mycétomateuse chez un patient immunodéprimé. J. Mycol. méd., 4:111-114, 1994. [ Links ]
5. DUPONT, B.; IMPROVISI, L. & RONIN, O. Aspects épidémiologiques et cliniques des infections à Scedosporium et à Pseudallescheria. J. Mycol.méd., 118:33-42, 1991. [ Links ]
6. DWORZACK, D.L.; CLARK, R.B.; BORKOWSKI, W.J. et al. Pseudallescheria boydii brain abscess: association with neardrowning and efficacy of high-dose, prolonged miconazole therapy in patients with multiple abscesses. Medicine (Baltimore), 68:218-224, 1989. [ Links ]
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9. HOOG, G.S. de; MARVIN-SIKKEMA, F.D.; LAHPOOR, G.A. et al. Ecology and physiology of the emerging opportunistic fungi Pseudallescheria boydii and Scedosporium prolificans. Mycoses, 37:71-78, 1994. [ Links ]
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11. LOPES, J.O.; ALVES, S.H.; BENEVENGA, J.P. et al. Subcutaneous pseudallescheriasis in a renal transplant recipient. Mycopathologia (Den Haag), 125:153-156, 1994. [ Links ]
12. LUTWICK, L.I.; RYTEL, M.W.; YANEZ, J.P. et al. Deep infections from Petriellidium boydii treated with miconazole. J. Amer. med. Ass., 241:272-273, 1979. [ Links ]
13. MADER, J.T.; REAM, R.S. & HEATH, P.W. Petriellidium boydii (Allescheria boydii) sphenoidal sinuditis. J. Amer. med. Ass., 239:2368-2369, 1978. [ Links ]
14. MENT, H.S.; SMITH, R.R.; KARP, J.E. et al. Pulmonary, cardiac and thyroid involvement in disseminated Pseudallescheria boydii. Arch. Path. Lab. Med., 108:859-861, 1984. [ Links ]
15. MILLER, M.A.; GREENBERGER, P.A.; AMERIAN, R. et al. Allergic bronchopulmonary mycosis caused by Pseudallescheria boydii. Amer. Rev. resp. Dis., 148:810-812, 1993.
16. MILNE, L.J.R.; McKERROW, W.S.; PATERSON, W.D. et al. Pseudallescheriasis in northern Britain. J. Med. vet. Mycol., 24:377-382, 1986. [ Links ]
17. MORIN, O.; DAUPHIN, B.; AUDOUIN, A.F. et al. Mycose sous-cutanée extensive à Scedosporium apiospermum chez un malade immunodéprimé. Étude clinique et épidémiologique. Bull. Soc. franç. Mycol. méd., 16:351-356, 1987. [ Links ]
18. PELLUFO, I.C.; CONTI-DIAZ, I.A.; CIVILA, E. et al. Micetoma por Scedosporium apiospermum (Petriellidium boydii) asociado a nocardiosis pulmonar, candidosis y dermatofitosis en un paciente immuno-deprimido. Dermatologia, 22:41-47, 1978. [ Links ]
19. PIENS, M.A.; JIMENEZ, J.L.; GUYOTAT, D. et al. A propos de trois observations dinfection humaine à Scedosporium apiospermum. Intérêt du traitement par itraconazole. J. Mycol. méd., 1:156-159, 1991. [ Links ]
20. RABODONIRINA, M.; PAULUS, S.; THEVENET, F. et al. Disseminated Scedosporium prolificans (S. inflatum) infection after a single lung transplantation. Clin. infect. Dis., 19:138-142, 1994. [ Links ]
21. RIPPON, J.W. Medical Mycology. The pathogenic fungi and the pathogenic actinomycetes. 3. ed. Philadelphia, W.B. Saunders, 1988. p. 651-680. [ Links ]
22. SCHERR, G.R.; EVANS, S.G.; KIYABU, M.T. et al. Pseudallescheria boydii infection in acquired immunodeficiency syndrome. Arch. Path. Lab. Med., 116:535-536, 1992. [ Links ]
23. SMITH, A.G.; CRAIN, S.M.; DEJONGH, C. et al. Systemic pseudoallescheriasis in a patient with acute myelocytic leukemia. Mycopathologia (Den Haag), 90:85-89, 1985. [ Links ]
24. TRAORE, F.; BEDROSSIAN, J. & BADILLET, G. Scedosporiose disseminé avec aneurismes chez un transplant renal. J. Med. Mycol., 1:149-151, 1991. [ Links ]
25. TRAVIS, L.B.; ROBERTS, G.D. & WILSON, W. R. Clinical significance of Pseudalescheria boydii: a review of 10 years experience. Mayo Clin. Proc., 60:531-537, 1985. [ Links ]
26. Van HECKE, E.; GEERTS, M.L. & Den DOOVEN, D. Petriellidiosis of the skin. Mykosen, 26:17-21, 1982. [ Links ]
27. VILLELA, J.M.; JACQUEMIN, J.L.; ROSSI, F. et al. Microabscés cutanés à Scedosporium apiospermum. Presse méd., 14:1516, 1985. [ Links ]
28. WILSON, C.M.; OROURKE, E.J.; McGINNIS, M.R. et al. Scedosporium inflatum: clinical spectrum of a newly recognized pathogen. J. infect. Dis., 161:102-107, 1990. [ Links ]
29. ZAFFIRO, A. Forma sigolare di mycosi cutanea do Monosporium apiospermum a sviluppo clinicamente setticemico. G. Med. milit., 86:636-640, 1938. [ Links ]
Recebido para publicação em 05/05/1997
Aceito para publicação em 10/09/1997
Correspondence to: Dr. Luiz Carlos Severo. IPD-Santa Casa, Annes Dias 285, 90020-090 Porto Alegre, RS, Brasil. Fax: 5551228-1566 r.3604, E-mail: email@example.com