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Subcutaneous phaeohyphomycosis
Abstract
We report the case of a 13-year-old boy who presented with multiple swellings all over the body. His condition remained undiagnosed for over 3 years. Exophiala spinifera was recovered from pus drained from the swellings. We discuss the difficulties in the initial diagnosis and the case of correct diagnosis once we had used special fungal stains.
... The isolated fungus was Exophiala spinifera in five separate case reports. [3][4][5][6][7] The other reported fungal isolates were Chaetomium globosum, 8 Veronaea botryose, 9 and Alternaria. 10 To the best of our knowledge, this is the first reported case of phaeohyphomycosis caused by Exophiala jeanselmei affecting an immunocompetent child. ...
... They occur more commonly in adults and are characterized by an acute onset of up to hundreds of dome-shaped pink to brown papules often on the trunk and extremities. 4,5 The scalp, mucous membranes, palms, and soles are typically spared, though in our patient the palms and soles were involved. The cause of EDSN is unknown, although a number of possible precipitating factors have been described in the literature, including pregnancy, 6 ultraviolet light exposure, 7 and fever following tonsillectomy. ...
... The cause of EDSN is unknown, although a number of possible precipitating factors have been described in the literature, including pregnancy, 6 ultraviolet light exposure, 7 and fever following tonsillectomy. 4 A Medline review yielded no previous reports in the literature of EDSN in an African-American. ...
... Five of the nine adults were immunosuppressed, and all suppressive regimens included prednisone or prednisolone in doses from 2.5 to 16 mg/day. One adult patient who was not on an immunosuppressive regimen during infection had a remote history of oral corticosteroids for asthma and her disease was exacerbated during pregnancy, a physiologically immunosuppressed state1415161718192021222324252627. Our case is the fifteenth case of clinical infection with E. spinifera reported in the English literature, and the first case to be definitively identified by molecular characterization. ...
... Importantly, it has been noted that in vitro susceptibility data seem to correlate poorly with in vivo efficacy [32]. Multiple modalities have been attempted to treat infection with E. spinifera, including the antifungals amphotericin B, ketoconazole, fluconazole, itraconazole, voriconazole, 5-fluorocytosine, terbinafine, griseofulvin, posaconazole, streptomycin, and the physical modalities heat, cryosurgery and excision [8,14151617181920212223242526. However, as detailed below, few of these treatments were ultimately found to be effective. ...
... Seven of the twelve patients who recovered from their disease, including our patient, were receiving itraconazole as part of their treatment regimen, and five were taking this medication as monotherapy. Four patients received amphotericin B in their initial treatment regimens; however, two patients required additional antifungal agents to obtain an adequate clinical response and one patient died, while one showed early improvement in short-term follow-up15161722]. Two cases reported increasing resistance to itraconazole over the course of treatment that was verified by in vitro testing [15,25], and one report noted decreasing efficacy of multiple antifungal treatments over a prolonged course [22], suggesting that E. spinifera possesses the ability to develop resistance to antifungal therapy. ...
Exophiala spinifera has been reported as an agent of cutaneous disease 18 times in the literature. Clinical presentations of cutaneous lesions vary widely, including erythematous papules, verrucous plaques, and deep subcutaneous abscesses. The clinical distribution and course of disease are also variable, depending on the age and immune competency of the patient. Histologic appearance occurs in one of two patterns--phaeohyphomycosis or chromoblastomycosis. While E. spinifera appears to be susceptible to multiple antimicrobial agents in vitro, clinical experience with treatment modalities has been variable. Prior to the availability of sequencing methods, species identification was based on the histopathologic presentation in tissue and morphologic features of the fungus in culture. It is likely that E. spinifera cutaneous infections have been underreported due to its incorrect identification based on earlier methods. We report an additional case of E. spinifera phaeohyphomycosis, the first to be definitively identified by sequencing. In addition, we summarize the variable clinical, histopathologic, and morphologic features, as well as treatment responses described in previously reported cutaneous infections caused by E. spinifera.
... Exophiala spinifera es un agente frecuente de feohifomicosis [32,37,43,52,[95][96][97][98][99][100] y algo menos de cromoblastomicosis [101,102]. Esporádicamente se ha referido como causa de infección diseminada [103][104][105] y queratomicosis [106]. ...
... En el tratamiento de la feohifomicosis han sido utilizados con éxito antifúngicos tales como itraconazol [37,39,40,51,54,57,58,85,97], anfotericina B [99], sola o asociada con ketoconazol [32,43] o 5-fluorocitosina [98], y ketoconazol combinado con 5-fluorocitosina [95]. ...
... Some cases (Rajam et al., 1958;Barba-Gomez et al., 1992;Padhye et al., 1996;Develoux et al., 2006;Tomson et al., 2006;Srinivas et al., 2016) were reported as chromoblastomycosis, although typical muriform cells were mostly lacking. Fatal systemic infections are known; 36 cases have been recorded in English and Chinese literature (Rajam et al., 1958;Nishimura and Miyaji, 1983;Padhye et al., 1983Padhye et al., , 1984Mirza et al., 1993;Campos-Takaki and Jardim, 1994;de Hoog et al., 1999;Rajendran et al., 2003;Negroni et al., 2004;Dutriaux et al., 2005;Baubion et al., 2008;Singal et al., 2008;Fothergill et al., 2009;Harris et al., 2009;Radhakrishnan et al., 2010;Li et al., 2011;Badali et al., 2012;Daboit et al., 2012;Lin et al., 2012;Bohelay et al., 2016;Silva et al., 2017; Table 2). Among the 12 systemic infections, all cases concerned immunocompetent patients. ...
Exophiala spinifera and Exophiala dermatitidis (Fungi: Chaetothyriales) are black yeast agents potentially causing disseminated infection in apparently healthy humans. They are the only Exophiala species producing extracellular polysaccharides around yeast cells. In order to gain understanding of eventual differences in intrinsic virulence of the species, their clinical profiles were compared and found to be different, suggesting pathogenic strategies rather than coincidental opportunism. Ecologically relevant factors were compared in a model set of strains of both species, and significant differences were found in clinical and environmental preferences, but virulence, tested in Galleria mellonella larvae, yielded nearly identical results. Virulence factors, i.e. melanin, capsule and muriform cells responded in opposite direction under hydrogen peroxide and temperature stress and thus were inconsistent with their hypothesized role in survival of phagocytosis. On the basis of physiological profiles, possible natural habitats of both species were extrapolated, which proved to be environmental rather than animal-associated. Using comparative genomic analyses we found differences in gene content related to lipid metabolism, cell wall modification and polysaccharide capsule production. Despite the fact that both species cause disseminated infections in apparently healthy humans, it is concluded that they are opportunists rather than pathogens.
... The influence of the age of the patient on the extensive nature of the lesions was also pointed out by de Hoog et al. (4) in infections caused by the phaeohyphomycotic agent Exophiala spinifera. The subcutaneous infections in children (8,10,13,14) involved extensive areas of the body, causing granulomatous crusty lesions. The infections in those cases became chronic and involved lymph nodes, severe osteomyelitis, and eventual fatal outcomes. ...
The second case of phaeohyphomycosis caused by Veronaea botryosa in China, in a 12-year-old boy from Jiangsu Province, is presented. Based on direct examination of the scrapings from crusted
lesions; histologic examination of the biopsy tissue showing septate, phaeoid hyphal elements; and the culture exhibiting
sympodial, conidiogenous cells producing predominantly two-celled, cylindric conidia, the etiologic agent was identified as
V. botryosa.
... 13 Subcutaneous abscesses involving the skin, muscle, and bone, associated with bone degeneration, have also been described in a patient from Brazil. 4 It is noteworthy that pheohyphomycosis caused by E. spinifera presents with single or a few lesions in immunocompromised adults, whereas multiple, widespread verrucous plaques occur in children with no evidence of immunosuppression. 5,7,10,12 Whether this is coincidental, or the result of the failure of development of specific immunity in the pediatric age group to limit the infection, needs to be evaluated. Treatment outcome in pheohyphomycosis has been variable. ...
A 10-year-old immunocompetent boy presented with multiple, verrucous, disseminated pheohyphomycotic lesions caused by Exophiala spinifera. The patient was not responsive to combination antifungal therapy (itraconazole, terbinafine, fluconazole) and cryotherapy. As antifungal susceptibility is known to be variable for Exophiala spinifera, in vitro sensitivity testing is recommended before medical treatment. This article reviews, in brief, all cases documented so far in the English literature.
Exophiala spinifera is a rare dematiaceous fungus causing cutaneous, subcutaneous and disseminated phaeohyphomycosis (PHM). Standard antifungal therapy for PHM is still uncertain. Here, we report a case of a Chinese male with PHM caused by E. spinifera, who received significant clinical improvement after the treatment with oral itraconazole and terbinafine. With the aim of evaluating the antifungal therapy for PHM caused by E. spinifera, a detailed review was performed.
Exophiala spinifera is a dematiaceous fungus responsible for rare skin infections presenting as phaeohyphomycosis or chromoblastomycosis which has been primarily reported in tropical and subtropical areas (Asia, South and North America). We report the first case of E. spinifera phaeohyphomycosis in a European patient. The phaeohyphomycosis was limited to the skin, involving the finger of an immunocompromised patient presenting with a large B-cell lymphoma treated by R-mini-CHOP regimen. Remission was initially achieved by surgical excision; however, a local subcutaneous relapse required treatment with itraconazole. We performed a literature review of the 32 previously published cases of E. spinifera phaeohyphomycosis highlighting its clinical phenotype: disseminated infection with extracutaneous involvement and poor prognosis were reported in young patients, of whom some were recently associated with CARD9 mutations, whereas cases in older immunocompromised patients were limited to the skin and showed better prognosis. There is currently no standard treatment for E. spinifera phaeohyphomycosis; however, itraconazole, alone or in combination, allowed partial or complete response in 16 out of 20 cases.
Using ITS1-2 sequence comparison, fifteen strains of Exophiala spinifera were found to be nearly identical to the type strain, CBS 899.68. Phaeococcomyces exophialae, based on type strain CBS 668.76, constituted a separate subgroup at a short distance; the taxon may be maintained at the variety level. Members of both groups can be entirely yeast-like or possess well-differentiated conidiophores. Strains from human and animal infections all were identified as E. spinifera s. str. No teleomorph is known; affinities to related Capronia species are discussed. A number of environmental strains with E. spinifera-like morphology could as yet not be identified. Exophiala jeanselmei is found to be a close relative of E. spinifera. Physiological profiles for phenetic recognition of E. spinifera are provided.
A case of Exophiala spinifera nasal infection is described in an 8-year-old female Birman cat who was examined for a nodulous mass protruding from the right nostril. It is the first clinical case record of E. spinifera from Europe and the second report of that black yeast infection in animals. Diagnosis was confirmed by histology, direct microscopic examination of biopsy samples and culture. Surgery in combination with a long-term permanent antifungal therapy with various drugs (oral ketoconazole [KTC] 10 mg kg-1 day-1 for 5 months, KTC and local enilconazole for 2 months, then fluconazole 10 mg kg-1 day-1 during 15 months) could not bring the cure. The cat was feline leukaemia virus antigen negative, feline immunodeficiency virus and feline infectious peritonitis virus antibodies negative; no predisposing factors could be found. Identical fungal elements were found in tissues before any specific treatment and after seven months of KTC therapy which did not prevent the isolation of E. spinifera in two occasions. Clinical aspect, aetiology, diagnosis, epidemiological data and treatment of the case are then discussed in a literature review about feline phaeohyphomycoses. This review concerns 37 feline cases of phaeohyphomycosis and phaeomycotic mycetoma that have been caused by at least 14 different species belonging to 12 genera of dematiaceous fungi.
We report a case of mucocutaneous phaeohyphomycosis caused by Exophiala spinifera. Crusty plaques and nodules were major clinical features. Histological examination revealed brown yeast-like cells and hyphae. Mycological and molecular data identified E. spinifera as etiologic agent. Oral itraconazole was effective, which was in accordance with the results of in vitro susceptibility testing. We speculated that her pregnancy may play a role of risk factor in the infection by E. spinifera.
The antifungal activities of miconazole, terbinafine, itraconazole, UR 9862, voriconazole and amphotericin B against 13 clinical isolates of Ochroconis gallopava were tested by broth microdilution methods. All drugs were very active against O. gallopava, MIC90 ranging between 0.06 and 2. Terbinafine and voriconazole seem to be promising agents in the treatment of infections caused by O. gallopava.
The second case of phaeohyphomycosis due to Exophiala spinifera in India has been diagnosed 46 years after the initial case. The present case involved a 12-year-old female patient with no known immunocompromising conditions. She presented with multiple verrucous, well-defined plaques encompassing phaeohyphomycotic lesions of varying sizes on her face, chest, arms and thighs. Lymph node involvement in dissemination was confirmed by demonstrating pigmented fungal elements in histopathology of the left axillary node. The infection responded positively to prolonged administration of itraconazole. The original case involved a young boy and was similarly disseminated but was more severe, with bone involvement, and had a fatal outcome. It is likely that other such cases have occurred in the intervening time but have not been reported.
Exophiala spinifera is a rare fungus causing chromoblastomycosis or different types of phaeohyphomycosis (cutaneous, subcutaneous, disseminated and cyst phaeohyphomycosis). We report a case of a young male with phaeohyphomycosis due to E. spinifera, who had multiple itchy painful papular lesions disfiguring his face for 4 years. His diagnosis was delayed and had received antibacterial and antileishmanial therapy elsewhere without any improvement. While he reported to our hospital, the histopathology of the biopsy collected from the lesion demonstrated acute on chronic inflammation with granuloma formation and darkly pigmented fungal elements. The isolate grown on culture was identified as E. spinifera on the basis of morphological characters. The identification of the isolate was further confirmed by sequencing of the ITS region of ribosomal DNA. After treatment with oral itraconazole, he had marked clinical improvement.
Here we report a case of a 55-year-old Indian male presenting with multiple subcutaneous cysts, which developed from painful nodules at the dorsal right wrist joint. Subsequently a painful nodule appeared on the left knee joint. Cytological examination of the knee swelling revealed a suppurative inflammatory lesion consisting of neutrophils, lymphocytes, multinucleated giant cells and few fungal elements, without involvement of the overlying skin. Exophiala spinifera was cultured (CBS 125607) and its identity was confirmed by sequencing of the internal transcribed spacer (ITS rDNA). The cysts were excised surgically, without need of additional antifungal therapy. There was no relapse during one-year follow-up and the patient was cured successfully. In vitro antifungal susceptibility testing showed that posaconazole (0.063 μg/ml) and itraconazole (0.125 μg/ml) had the highest and caspofungin (4 μg/ml) and anidulafungin (2 μg/ml) the lowest activity against this isolate. However, their clinical effectiveness in the treatment of E. spinifera infections remains to be evaluated. In this case report, we have also compiled cases of human E. spinifera mycoses which have been reported so far.
During the past four decades, seven patients were documented in China to have died from Exophiala infections. Causative agents were Exophiala dermatitidis, Exophiala spinifera, Exophiala jeanselmei and a new Exophiala species, Exophiala asiatica. We retrospectively analysed the clinical characteristics of these infections in China and confirmed the identity of aetiological agents of Chinese fatal cases using rDNA ITS sequence analysis. While E. dermatitidis displayed neurotropism, E. spinifera showed osteotropism. The other two species, E. jeanselmei and E. asiatica had caused brain infections in China.
Fungal infection of the skin and subcutaneous tissue may result from either direct contact or inoculation injury (primary infection) or from hematogenous spread from a primary focus of disease (secondary infection). The parainfectious lesions of erythema nodosum and erythema multiforme are manifestations of the host's immune response to the invading fungus, particularly Histoplasma capsulatum and Coccidiodes immitis. In some patients, skin lesions may be the only sign of a systemic fungal infection, and prompt recognition of these lesions may facilitate early diagnosis and treatment. This article first addresses the pathogenesis, host defenses, and diagnosis of fungal skin infections. The specific cutaneous manifestations of the superficial, cutaneous, subcutaneous, and systemic mycoses are then reviewed.
We report the second case of chromoblastomycosis caused by Exophiala spinijera; this is the first known case in the United States. Examination of biopsied tissue showed thick-walled, internally septated,
chestnut brown muriform cells (sclerotic bodies) within multinucleated giant cells present in the dermis that were characteristic
of chromoblastomycosis. The individual cells within the muriform cells disarticulated from the outer wall of the parent cell
and from each other to form endoconidia within the outer walls of the parent cells. After fracture of the outer walls, the
endoconidia were released. This unique process of endoconidial formation in vivo for the propagation of muriform cells was
observed for the first time. Initial treatment with itraconazole and 5-fluorocytosine followed by treatment with itraconazole
and heat resulted in marked improvement in the patient's lesions. This infection reiterates the fact that the dematiaceous
fungus E. spinijera, a well-known etiologic agent of phaeohyphomycosis, can cause more than one type of infection and supports earlier observations
that chromoblastomycosis and phaeohyphomycosis represent extremes of a continuum of infections.
Phaeohyphomycosis and nocardiosis are uncommon infections that are more frequently reported in immunocompromised patients. To our knowledge, this is the first report of subcutaneous phaeohyphomycosis caused by Exophiala jeanselmei in association with systemic infection with Nocardia asteroides . The patient's phaeohyphomycosis responded to surgical excision and multi-drug therapy, and the patient underwent prolonged therapy with trimethoprim/sulfamethoxazole for treatment of the nocardiosis. (J Am Acad Dermatol 1997;36:863-6.)
Phaeohyphomycosis describes a heterogenous group of mycotic infections caused by pigmented fungi. Previously uncommon to the United States, the number of case reports in the American literature has steadily increased over the past two decades. This has been attributed to the ever increasing number of immunocompromised individuals as well as an influx of immigrants from areas where these opportunistic fungi are more commonly found. The authors present a generalized overview of phaeohyphomycosis as well as a more specific breakdown and case study involving subcutaneous phaeohyphomycosis.
Ramichloridium obovoideum (“Ramichloridium makenziei”) is a rare cause of lethal cerebral phaeohyphomycosis. It has been, so far, geographically restricted to the Middle East.
BALB/c mice were inoculated with two strains ofR. obovoideum intracranially. Therapy with amphotericin B, itraconazole, or the investigational triazole SCH 56592 was conducted for 10
days. Half the mice were monitored for survival and half were killed for determination of the fungal load in brain tissue.
Recipients of SCH 56592 had significantly prolonged survival and lower brain fungal burden, and this result was found for
mice infected with both of the fungal strains tested. Itraconazole reduced the brain fungal load in mice infected with one
strain but not the other, while amphotericin B had no effect on brain fungal concentrations. This study indicates a possible
role of SCH 56592 in the treatment of the serious cerebral phaeohyphomycosis due to R. obovoideum.
The antifungal activities of miconazole, terbinafine, itraconazole, UR 9825, voriconazole and amphotericin B against 11 clinical isolates of Exophiala spp. were tested by the broth microdilution method. All drugs were very active against Exophiala spp.. The 90% minimal inhibitory concentration (MIC90) ranged from 0.125 to 1 microgram ml-1. Terbinafine was the most active drug against Exophiala spinifera, Exophiala dermatitidis and Exophiala castellanii and seems to be a promising agent in the treatment of infections caused by these fungi.
An immunosuppressed patient who presented with unusual clinical signs of cutaneous alternariosis, including papular, nodular and verrucous lesions of the forearms, is reported. In spite of continuous treatment with oral itraconazole for 6 months, a large, progressive, necrotic ulcer appeared on the patient's left leg. Liposomal amphotericin B was then administered (total dose, 750 mg) with excellent clinical results. Described here is the case of a renal transplant recipient with cutaneous and subcutaneous alternariosis, affecting both of the forearms and the left leg, with the clinical presentation differing according to location. The clinical response to long-term itraconazole therapy was poor, but treatment with liposomal amphotericin B was successful.
The pathogenicity of several dematiaceous yeasts that have, to date, rarely been isolated in humans remains unclear. Because professional phagocytes are prominent in lesions caused by dematiaceous fungi, we address this issue by comparing phagocytosis, evoked oxidative burst and killing by human neutrophils of different black yeasts in vitro. Whereas phagocytosis of all black yeasts tested and evoked oxidative burst yielded comparable results, in contrast, the degree of killing differed significantly after 5 h. Thereby, two groups could be identified; one in which strains are killed at high rates, for example, Hortaea werneckii (81 +/- 11.6%), Exophiala castellanii (96 +/- 8.6%), Phaeoannellomyces elegans (93 +/- 9.7%), Phaeococcomyces exophialae (87 +/- 8.7%), and the other in which strains are killed to a lesser degree, for example, Exophiala dermatitidis (ATCC 34100) (61 +/- 9.5%), E. dermatitidis (CBS 207.35) (66 +/- 7.5%), E. jeanselmei (50 +/- 10.5%), E. mesophila (63 +/- 11.6%), E. bergeri (63 +/- 9.1%), and E. spinifera (57 +/- 9.6%). Non-pigmented yeasts were killed at levels comparable with those at which the white mutant strain of E. dermatitidis (ATCC 44504) was killed (95 +/- 7.5%); the yeast strains tested were Candida albicans (DSM 11943) (95 +/- 4.0% killing) and Saccharomyces cerevisiae (DSM 1333) (95 +/- 10.3%). Comparison of killing rates with the observed pathogenicity of the melanized species suggests that low killing rates might indicate or even predict a high degree of invasiveness. Although previous experiments revealed that melanization conferred killing resistance on E. dermatitidis, the differences in killing rates of other dematious fungi suggest that melanization of the cell wall is in itself insufficient to confer virulence.
Clinical and laboratory aspects of the infections caused by Exophiala species are reviewed with regard to its epidemiology, diagnosis and treatment. Exophiala is a genus of dematiaceous hyphomycetes whose taxonomy and nomenclature undergo constant revision. Exophiala species are widely distributed in nature, and they are uncommon human pathogens. In recent years it appears to have increased its frequency as a cause of human infections, mainly in immunocompromised patients. They have been associated with phaeohyphomycosis, chromoblastomycosis, eumycotic mycetoma and disseminated infection. The procedures recommended for diagnosis consist of detection of fungal elements in tissue and growth of the organism in culture. Identification is mostly based upon microscopic observation of morphological characteristics and conidiogenesis, combined with the evaluation of physiological tests and nitrate and carbohydrate assimilations. Antifungal agents such as amphotericin B, itraconazole and voriconazole showed in vitro activity to most of the Exophiala species of clinical interest. The therapeutic recommendations are mainly deduced from the observation of single cases.
Antifungal susceptibility profiles were determined for 16 strains of the black yeast Exophiala spinifera applying different temperature regimens. Fluconazole was the least effective in vitro. Lowest minimal inhibitory concentration (MIC) values were found with itraconazole. The activities of antifungal agents against environmental and clinical strains were similar. Post-antifungal effect (PAFE) of four drugs was determined for 11 clinical strains. PAFE was observed only for amphotericin B, with extended inhibition times seen at high drug concentrations.
The second case of phaeohyphomycosis due to Exophiala spinifera in India has been diagnosed 46 years after the initial case. The present case involved a 12-year-old female patient with no known immunocompromising conditions. She presented with multiple verrucous, well-defined plaques encompassing phaeohyphomycotic lesions of varying sizes on her face, chest, arms and thighs. Lymph node involvement in dissemination was confirmed by demonstrating pigmented fungal elements in histopathology of the left axillary node. The infection responded positively to prolonged administration of itraconazole. The original case involved a young boy and was similarly disseminated but was more severe, with bone involvement, and had a fatal outcome. It is likely that other such cases have occurred in the intervening time but have not been reported.
The four varieties of human infections caused by fungi whose mycelium or conidia or both are dark colored, are described and discussed under the headings of superficial, cutaneous, subcutaneous and systemic mycoses. Three diseases, black piedra, tinea nigra and mycotic keratitis, were considered in the superficial category. Their etiologic agents, respectively 4 and 17 in number were briefly described in terms of their clinical manifestations, in vivo and in vitro morphology. The subcutaneous mycoses with dematiaceous etiologic agents are chromoblastomycosis and phaeohyphomycosis. The former is considered to be caused by any one of six moulds classified in the genera Cladophialophora, Cladosporium, Fonsecaea, Phialophora and Rhinocladiella. The fungi incriminated as agents of phaeohyphomycosis, currently total 32. The fundamental differences between chromoblastomycosis and phaeohyphomycosis lies in the tissue form assumed by their respective etiologic agents. In chromoblastomycosis, the parasites occur as large, muriform, thick-walled dematiaceous cells. In phaeohyphomycosis the fungi basically occur as dark walled septate, hyphal elements. The mycetomas with black granules are known to be caused by 9 species of moulds classified in 6 genera. Within limits, the identity of a given black grained mycetoma can be deduced through the characteristics of the granules.
The authors describe a subcutaneous abscess on the arm of a coloured Senegalese caused by a dematiaceous fungus. In its parasitic state, the fungus appears in the form of filaments and unicellular or budding brownish yellow elements. The macro- and microscopical features were studied in culture and attention directed towards certain morphological characteristics of the sporing structures and the mode of formation of the spores. After critical examination of the evidence encountered in the literature, it was possible to identify the causal fungus as Phialophora gougerotii. The taxonomical status of this genus is confused but at the moment it would appear that the name Phialophora is the most appropriate.
The authors propose the term “phaeo-sporotrichosis” for all mycotic abscesses induced by dematiaceous fungi belonging to these genera and species which although often very different the parasitic phase is always brown and filamentous and more or less fragmented.
They therefore regard the naming of “sporotrichosis” (sic) when applied to some of these mycoses as traditional but erroneous.
A previously undescribed host for the opportunistic dematiaceous hyphomycete, Scolecobasidium humicola, is reported. Several epizootics among rainbow trout, Salmo gairdneri, occurred in a Tennessee fish hatchery from 1969 to 1973. Symptoms included surface lesions, blisters and abscesses. The kidneys and other internal organs were invaded by the mycelium of S. humicola. Tissue morphology of the fungus was typical of that associated with phaeohyphomycosis Experimental infections were reproduced in fingerling rainbow trout after intraperitoneal inoculation of S. humicola. Following a change in the hatchery's water supply, no new epizootics have occurred.
Cladosporium trichoides was isolated from brain abscesses of two cats. No obvious route of entry was found in either case. The brain was the only organ cultured. No other significant lesions were observed during necropsy. These are the first reported cases of such infection in animals.
A slowly evolving subcutaneous mycosis in a 10-year-old domestic shorthair cat was found to be caused by Drechslera spicifera, the imperfect state of ascomycete Cochliobolus spicifer. The cat had circular, nodular, granulomatous lesions over its sternum. Scattered individual and small groups of septate hyphae and chlamydospores were found in histologic sections. Many of the hyphae also had bizarre dilatations. Most of the fungal elements were hyaline; a few, however, were dematiacious. Because the fungus was not organized into granules in tissue, the disease could not be classified as a mycetoma. The preferred name for infections of this type is phaeohyphomycosis.
The results of long-term itraconazole therapy in 10 patients with active chromoblastomycosis due to F. pedrosoi were reported. Therapy consisted of 100 or 200 mg/day of itraconazole, the length of therapy depending on the patient's response (12 to 24 months). This new triazole proved effective in reducing the number, size, and severity of the lesions in nine of the patients. Those patients with minor involvement profited more from therapy and were cured; patients with moderate involvement achieved either minor or major improvement. In most cases, signs and symptoms began to improve after 6 months of therapy. Mycological tests (in which tissue samples were treated with potassium hydroxide and cultured) became negative in six patients, but the fungus was eradicated in only three patients. Itraconazole produced no side effects. In spite of the need for long-term therapy, this new azole derivative effectively controls the disease.
Itraconazole has been used as oral therapy for deep mycoses since July 1984 in our institution. So far, therapy has been evaluated
for eight patients: one with cutaneous coccidioidomycosis; two with chromomycosis (Fonsecaea pedrosoi); and five with sporotrichosis (three fixed, one Iymphangitic, and one disseminated). Clinical and mycologic evaluations were
done at the beginning of treatment, 15 days after the initiation of treatment, and monthly thereafter. Dosage was 100–200
mg per day, and duration of treatment was based on response. The cases of coccidioidomycosis and lymphangitic sporotrichosis
were mycologically cured after two and seven months of treatment with 100 mg per day, respectively. The six other patients
required higher doses. Two patients with sporotrichosis and one with chromomycosis were cured, and one patient with sporotrichosis
and one with chromomycosis were markedly improved. In one patient with sporotrichosis, treatment was discontinued because
of the slow response. No adverse reactions to treatment were reported.
Itraconazole was administered orally to two patients with sporotrichosis, 10 patients with paracoccidioidomycosis, three with
mycetomas (due to Madurella grisea, Streptomyces madurae, and Pseudochaetosphaeronema larense, respectively), nine with chromomycosis due to Cladosporium carrionii, five with chromomycosis due to Fonsecaea pedrosoi and five with leishmaniasis (including one with the nodular disseminated form). The clinical and laboratory tests showed
excellent tolerance to the drug with a total absence of adverse reactions. Satisfactory results were achieved against paracoccidioidomycosis,
sporotrichosis, and chromomycosis due to C. carrionii (apparent cure was achieved in a short time). Encouraging improvement was noted in the treatment of mycetoma due to M. grisea. Among the five cases of leishmaniasis, a complete clearing was achieved in one and an encouraging improvement in two, including
the one with the nodular disseminated form. Two patients with F. pedrosoi infection were apparently cured after the addition of thermotherapy and flucytosine, respectively, to the treatment regimen.
A subcutaneous fungus infection that developed in a kidney transplant patient on immunosuppressive maintenance therapy was found to be caused by a new species of Phialophora. This species, P. parasitica, developed in the host's tissues in the form of dematiaceous mycelium. In culture on Sabouraud dextrose agar it is characterized by the formation of phialides that extrude ovoid to ellipsoid phialospores at maturity. The term phaeohyphomycosis is proposed as a collective name for a group of mycoses caused by diverse genera and species of dematiaceous fungi. All the etiologic agents, despite their taxonomic heterogeneity, are characterized and united by the development of dark mycelial elements in the host's tissues. The term 'phaeosporotrichosis,' previously introduced by others for this type of disease, should be discarded on the grounds of inappropriateness.
This is a case report of a rare cerebellar abscess from which the dematiaceous fungus C. trichoides (bantianum) was isolated and tentatively identified during the patient's lifetime. Involvement of the posterior fossa has been reported in only 2 other recorded cases of cladosporiosis. As is common in such cases, the portal of entry could not be determined. Despite excision of the abscess and the institution of treatment with amphotericin B, the patient died 10 days postoperatively. There is no known effective cure for this disease.
A fatal encephalitis of chickens, caused by the thermophilic fungus Dactylaria gallopava, affected over 200 birds in a flock of 65,000 broilers. The disease was reproduced experimentally by inoculating spore suspensions into 1 day old chicks via the left posterior thoracic air sac, the left maxillary sinus, and also intracerebrally. Gross and microscopic lesions were found in the brains, air sacs, lungs, eyes, and livers. The brain lesions were like those in the natural outbreak, and D. gallopava was recovered from the inoculated chickens. The brain lesions were compared with those in birds with aspergillosis.
From an unusual verrucous lesion on the face of an Indonesian boy we repeatedly isolated a fungus which has not been reported previously as a cause of disease in man or animals. The fungus obviously falls within the taxonomic limits of the genus Cercospora, whose many species are well known to plant pathologists as frequent causes of leaf spots. We are unable to differentiate the fungus from Cercospora apii Fresenius 1863, and we identify it with this species. The production of extensive dermal lesions in man with invasion and proliferation of hyphae to the corium by a fungus which is a known pathogen of plants is unique in medicine.
This case will be reviewed, along with a second unusual subcutaneous mycosis,4 in a paper prepared for a mycological journal.2
Report of Case
An Indonesian orphan boy, age 12 years, was hospitalized Aug. 28, 1954, because
Feline abscess due to Cladosporium trichoidis
- Jang
- Ss
- Rinaldi El Mg Biberstein
- Am
- Boorman
- Ga
- Taylor
Jang SS, Biberstein EL, Rinaldi MG, Henness AM, Boorman GA, Taylor RF. Feline abscess due to Cladosporium trichoidis. Sabouraudia. I977; 15: I15-x23.
Phaeohyphomycosis : definition and etiology. In: Mycosis, Scientific publication No. 3o 4
- L Ajello
Ajello L. Phaeohyphomycosis : definition and etiology. In: Mycosis, Scientific publication No. 3o 4. Washington, DC: Pan American Health Organisation, I975: I26-I33.
Treatment of chromo-blastomycoses with itraconazole I2. BoreUi D. A clinical trialof itraconazole in the treatment of deep mycoses and leishmaniasis
- I I A Restrepo
- A Gonzalez
- I Gomez
- M Arango
- Bedout
I I. Restrepo A, Gonzalez A, Gomez I, Arango M, de Bedout C. Treatment of chromo-blastomycoses with itraconazole. Ann N Y Acad Sci I988; 544:5o4-5 I6. I2. BoreUi D. A clinical trialof itraconazole in the treatment of deep mycoses and leishmaniasis. Rev Infect Dis 1987; 9 (Suppl I): s57-63.
Kyste souscutan6e mycosique (phaeosporotricose) ~ Phialophora gougerotii
- F Mariat
- Destombes G P Segretain
- Daresse
Mariat F, Segretain G, Destombes P, Daresse H. Kyste souscutan6e mycosique (phaeosporotricose) ~ Phialophora gougerotii (Matruchot 19Io, Borelli 1955) observ6 au Senegal Sabouraudia I967; 5: 2o9-219.