Generalized Subcutaneous Phaeohyphomycosis Caused by Phialophora verrucosa: Report of a Case and Review of Literature

We report a case of subcutaneous phaeohyphomycosis due to Phialophora verrucosa in a 64-year-old Chinese farmer suffering from CD4+ lymphopenia. He presented with diffuse and infiltrated plaques involving the entire face including the eyes, neck, occiput, and extending to the dorsal regions of his torso. The patient is notable for the discrete multifocal nature of the illness in the absence of disseminated infection and rarity of P. verrucosa as a cause of subcutaneous phaeohyphomycosis.     

Subcutaneous cystic granuloma caused byPhialophora verrucosa

A case of subcutaneous cystic granuloma caused byPhialophora verrucosa was reported. The patient was 21-year-old Japanese female with systemic lupus erythematosus (SLE) who had received prolonged steroid therapy. P. verrucosa has occasionally been isolated from patients as one of etiological agents of chromomycosis. There have been, however, no paper published to report an isolation of this fungus from subcutaneous cyst or abscess of a patient. This case is the first report of subcutaneous cystic granuloma induced clinically byP. verrucosa.     

Successful Posaconazole Therapy of Disseminated Alternariosis due to <Emphasis Type="Italic">Alternaria infectoria</Emphasis> in a Heart Transplant Recipient

We report a case of phaeohyphomycosis caused by Alternaria infectoria in a 61-year-old heart transplant recipient with multiple skin lesions and pulmonary infiltrates. The infection spread via the haematogenous route from the primary cutaneous lesions into the lungs. The diagnosis was based on the histopathological examination, direct microscopy, skin lesion cultures and detection of Alternaria DNA in the bronchoalveolar lavage fluid using molecular methods. The treatment consisted of a combination of surgical excision and systemic antifungal therapy. Voriconazole was the first agent used but had a weak effect. Posaconazole was subsequently used to achieve a successful response. The isolate was identified as A. infectoria by sequencing of the rDNA ITS region and the partial β-tubulin gene.     

Phialophora verrucosa-induced subcutaneous phaeohyphomycosis. Fine needle aspiration findings

A 34-year-old woman on immunosuppressive therapy presented with a subcutaneous, cystic lesion on the dorsum of the right foot. Cytologic examination of material obtained by fine needle aspiration (FNA) revealed a mixture of acute and granulomatous inflammation as well as brown-pigmented fungi in the form of budding yeast, pseudohyphae and septate hyphae. The findings suggested subcutaneous phaeohyphomycosis (phaeomycotic cyst). Culture grew Phialophora verrucosa. The cytologic, histologic and cultural findings are given. This case demonstrates that phaeohyphomycosis can be diagnosed by FNA but that fungal culture is necessary to establish the identity of the etiologic agent. This appears to be the first case of P. verrucosa-induced subcutaneous phaeohyphomycosis reported in the Western Hemisphere.     

Report of chronic subcutaneous abscesses caused byExophiala spinifera

A case of phaeohyphomycosis with subcutaneous abscesses involving skin, muscle and bone was caused byExophiala spinifera. This is the first report ofE. spinifera causing bone degeneration in Recife-PE, Brazil.     

Meanderella rijsii,a new opportunist in the fungal order Pleosporales

Subcutaneous phaeohyphomycosis is an implantation disease caused by melanized fungi and affect both immunocompetent as well as immunocompromised individuals. Diagnosis and treatment require proper isolation and accurate identification of the causative pathogen. We isolated a novel fungus from a case of subcutaneous phaeohyphomycosis in an immunocompetent patient. The 56-year-old patient suffered from a slowly progressive swelling on the metatarsophalangeal join of the left food. The isolated fungus lacked sporulation and sequences of the ribosomal operon did not match with any known species. In a multi-locus phylogenetic analysis involving five markers, the fungus formed a unique lineage in the order Pleosporales, family Trematosphaeriaceae. A new genus, Meanderella and a new species, Meanderella rijsii are here proposed to accommodate the clinical isolate. Whole genome analysis of M. rijsii revealed a number of genes that can be linked to pathogenicity and virulence. Further studies are however needed to understand the role of each gene in the pathogenic process and to determine the origin of pathogenicity in the family of Trematosphaeriaceae.     

Cutaneous infection by Phaeoacremonium parasiticum

BackgroundPhaeoacremonium parasiticum is considered a rare infectious agent that is part of a heterogeneous group of fungi causing phaeohyphomycosis. This organism is capable of producing subcutaneous infections, eumycetomas, osteomyelitis, arthritis, myositis and also disseminated diseases, such as fungemia and endocarditis.Case reportWe describe a case of cutaneous infection by P. parasiticum in a kidney transplant patient. The identification of this microorganism was performed by microbiological and histopathological studies and confirmed with the sequence of the gene encoding β-tubulin and a real time panfungal PCR targeting 18S ribosomal RNA gene. The microorganism was correctly identified by phenotypic and molecular methods. The patient was treated with oral antifungal therapy and a debulking surgery and evolved without any complication.ConclusionsThe diagnosis of this infection is difficult and usually affects kidney transplant patients, but the reasons of this association are still unknown.     

Spontaneous Remission of Subcutaneous Scedosporiosis Caused by <Emphasis Type="Italic">Scedosporium dehoogii</Emphasis> in a Psoriatic Patient

To date, only one case of post-traumatic endophthalmitis caused by Scedosporium dehoogii has been reported, but its contamination or colonization might not be precluded due to the absence of pathogenic isolation and/or pathological examination. We report the first case to our knowledge of S. dehoogii-induced subcutaneous scedosporiosis in a psoriatic patient. A 58-year-old man with 5-year history of psoriasis vulgaris and immunosuppressant therapy developed pyrexia and multiple subcutaneous abscesses on both knees. Direct microscopy of the yellowish pus showed masses of bright green short spores. Skin biopsy revealed some branched septate hyphae within the granuloma. Two aspirated pus specimens collected at a 1-week interval produced white cottony colonies on Sabouraud dextrose agar. Bacterial cultures of one blood and two purulent samples were negative, and fungal culture of blood sample was not performed. The isolate was identified as S. dehoogii using β-tubulin phylogeny and species-specific PCR with primer MSDE1/MSA2. Without addition of antifungal treatment, subcutaneous lesions disappeared spontaneously after immunosuppressant withdrawal and no relapse occurred during 64-month follow-up. The spontaneous recovery may result from immune reconstitution following immunosuppressant discontinuation.     

Congenital cutaneous candidiasis associated with maternal peripartum candidemia

BackgroundCutaneous congenital candidiasis (CCC) is a rare condition consisting of invasive fungal infection of the epidermis and dermis that mostly affects preterm infants. Maternal vaginal candidiasis is present in half of the cases, although the occurrence of invasive candidiasis during pregnancy or peripartum period is exceptional.Case reportWe present the case of a full-term infant that was born by vacuum-assisted vaginal delivery to an apparently healthy 33 year-old woman with no history of intravenous drug use or vaginal candidiasis during pregnancy. The newborn showed a diffuse maculopapular rash with respiratory distress and bilateral interstitial lung infiltrates, requiring nasal continuous positive airway pressure support. Blood cultures obtained from the mother due to intrapartum fever yielded Candida albicans. Cultures of vaginal discharge and neonate skin also yielded C. albicans with the same in vitro susceptibly pattern. No alternative source for candidemia was identified. The clinical course after starting a systemic antifungal therapy was favorable in both the mother and the neonate, with clearance of candidemia and resolution of the skin lesions.ConclusionsCCC must be considered in full-term newborns with maculopapular rash at birth or during the first days of life. The absence of alternative sources for bloodstream infection in the present case suggests a potential etiopathogenic relationship between CCC and maternal candidemia. It is reasonable to rule out postpartum candidemia when CCC is suspected.     

Cutaneous and Mucosal Phaeohyphomycosis Caused by Exophiala spinifera in a Pregnant Patient: Case Report and Literature Review

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.     

Subcutaneous Phaeohyphomycosis Caused by <Emphasis Type="Italic">Exophiala oligosperma</Emphasis> in an Immunocompetent Host: Case Report and Literature Review

We report a case of subcutaneous infection caused by Exophiala oligosperma. Erythematous ulcerated plaque with exudate was major clinical features. Histopathological examination showed yeast-like cells and fungal hyphae. Mycological and molecular identification revealed E. oligosperma as etiologic agent. Local debridement and oral itraconazole were effective. To the best of our knowledge, this is the first report of phaeohyphomycosis caused by E. oligosperma in mainland China. This report highlights the potential role of E. oligosperma as an emerging cause of infection in immunocompetent patients.     

Subcutaneous phaeohyphomycosis due to Curvularia lunata in a renal transplant patient

BackgroundPhaeohyphomycosis is defined as an infection caused by melanized fungi. It predominates in tropical climate and is currently classified as superficial, allergic, central nervous system or lung infections, and disseminated. Curvularia is one of the many genres which can cause this disease. Phialophora, Alternaria and Exophiala are more commonly isolated from subcutaneous lesions.Case reportA 25-year-old male, renal transplant recipient presents with an ulcer on his left leg. Subcutaneous phaeohyphomycosis due to Curvularia lunata was diagnosed based on mycological examination and histopathological findings. He was successfully treated with systemic itraconazole and surgical resection.ConclusionThe incidence of phaeohyphomycosis has increased in the last decades, especially in immunosuppressed individuals; nevertheless the number of cases does not allow for therapeutic controlled trials to be performed. Hence, we consider that it is important to communicate individual cases and reviews of the literature, to increase awareness of the disease, its clinical presentation and response to treatment.     

Altered Microbiomes in Bovine Digital Dermatitis Lesions,and the Gut as a Pathogen Reservoir

Bovine digital dermatitis (DD) is the most important infectious disease associated with lameness in cattle worldwide. Since the disease was first described in 1974, a series of Treponema species concurrent with other microbes have been identified in DD lesions, suggesting a polymicrobial etiology. However, the pathogenesis of DD and the source of the causative microbes remain unclear. Here we characterized the microbiomes of healthy skin and skin lesions in dairy cows affected with different stages of DD and investigated the gut microbiome as a potential reservoir for microbes associated with this disease. Discriminant analysis revealed that the microbiomes of healthy skin, active DD lesions (ulcerative and chronic ulcerative) and inactive DD lesions (healing and chronic proliferative) are completely distinct. Treponema denticola, Treponema maltophilum, Treponema medium, Treponema putidum, Treponema phagedenis and Treponema paraluiscuniculi were all found to be present in greater relative abundance in active DD lesions when compared with healthy skin and inactive DD lesions, and these same Treponema species were nearly ubiquitously present in rumen and fecal microbiomes. The relative abundance of Candidatus Amoebophilus asiaticus, a bacterium not previously reported in DD lesions, was increased in both active and inactive lesions when compared with healthy skin. In conclusion, our data support the concept that DD is a polymicrobial disease, with active DD lesions having a markedly distinct microbiome dominated by T. denticola, T. maltophilum, T. medium, T. putidum, T. phagedenis and T. paraluiscuniculi. Furthermore, these Treponema species are nearly ubiquitously found in rumen and fecal microbiomes, suggesting that the gut is an important reservoir of microbes involved in DD pathogenesis. Additionally, the bacterium Candidatus Amoebophilus asiaticus was highly abundant in active and inactive DD lesions.     

Phaeohyphomycosis caused by Cladophialophora bantiana

BackgroundCladophialophora bantiana is the most frequent cause of central nervous system phaeohyphomycosis.AimsWe report a case of phaeohyphomycosis by C. bantiana in a patient with underlying lung disease on steroid therapy.MethodsAn 81-year-old male was admitted in August 2011 with a history of difficulty speaking and deflection of the oral commeasure to the left side with a brain abscess. Brain tissue was cultured on Sabouraud media and sequence analysis of the internal transcribed spacer region of the ribosomal DNA was done for identification purposes. Susceptibility testing to various antifungal agents was performed using the microdilution test.ResultsHistopathological examination of the brain tissue ruled out malignancy and the presence of dematiaceous hyphae was observed. Culture showed the presence of a single black fungus, identified as C. bantiana. It was susceptible to all antifungals, except to caspofungin. The patient was treated with voriconazole plus liposomal amphotericin B. Cerebral cranial computed tomography [CCT] scans demonstrated persistence of the intraparenchymal abscess collection. Despite surgical and medical treatment with antifungal drugs, the patient died 5 months after the first diagnosis of the cerebral occupying lesion was made.ConclusionsPhaeohyphomycosis is an uncommon infection with severe limitations on the clinical clues that can help in early diagnosis. Fungal species identification is mandatory for epidemiological and therapeutic reasons. The MICs could be useful in selecting the appropriate antifungal agent. Avoiding the unnecessary exposure to soil or other media potentially contaminated with fungal spores should be recommended to any immunosuppressed patient.     

播散性暗色丝孢霉病患者CARD9突变及相关免疫学研究

暗色丝孢霉病是指由暗色真菌引起的皮肤、皮下组织乃至深部组织脏器的感染。本研究探讨1例由皮肤及皮下组织暗色丝孢霉病逐渐进展为播散性暗色丝孢霉病患者的遗传背景及其抗真菌免疫功能。收集患者10余年病情进展的临床资料及真菌检查结果,进行临床资料及真菌学研究;对外周血DNA采用Sanger测序,进行遗传学研究;提取患者及正常人外周血单个核细胞(peripheral blood mononuclear cell,PBMC),采用蛋白免疫印迹法、流式细胞术、化学发光法、酶联免疫吸附试验等进行免疫学研究。结果显示,患者胱天蛋白酶募集域蛋白9(caspase recruitment domain-containing protein 9,CARD9)基因存在新的复合杂合错义突变(p.R35Q和p.E81K),CARD9蛋白表达正常,外周血淋巴细胞(peripheral blood lymphocyte,PBL)Th1和Th17细胞比例均降低;PBMC抗疣状瓶霉感染存在天然及适应性免疫缺陷。本研究首次报道1例播散性暗色丝孢霉病患者存在CARD9基因新发错义突变,同时发现患者PBMC抗真菌的天然及适应性免疫存在缺陷,提示CARD9蛋白在暗色丝孢霉病发病中可能发挥重要作用。     

Chronic Disfiguring Facial Lesions in an Immunocompetent Patient Due to Exophiala spinifera: A Case Report and Review of Literature

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.     

Chromoblastomycosis due to Fonsecaea pedrosoi and F. monophora in Cuba

We report two cases of chromoblastomycosis due to Fonsecaea pedrosoi and F. monophora in otherwise healthy Cuban males. Direct microscopic examination of biopsies revealed muriform cells, the hallmark of chromoblastomycosis. The suspected agents were recovered in culture, identified on the basis of morphological criteria and confirmed by sequencing of the internal transcribed spacer regions of rDNA. Final treatment consisted of surgical excision. The patients were successfully cured since there was no relapse after a follow-up of more than a year. In vitro antifungal susceptibility testing of both isolates showed that itraconazole and posaconazole had potent activity. High MICs of amphotericin B (2 μg/ml), fluconazole (>64 μg/ml), anidulafungin (8 μg/ml) and caspofungin (8 μg/ml) were found.     

The First Case of Phaeohyphomycosis Caused by Rhinocladiella basitona in an Immunocompetent Child in China

We present the first case of phaeohyphomycosis caused by Rhinocladiella basitona (R. basitona) in China and describe the mycological characteristics of this pathogen. A 11-year-old girl was presented with plaque on her face for 3 years. Diagnosis was based on histopathology, mycology, and molecular identification. The patient was treated with terbinafine and itraconazole. This case is the second of phaeohyphomycosis caused by R. basitona in the world (previously belonging to Geniculosporium).     

Tissue changes in cryptococcosis: histologie alteration from gelatinous to suppurative granulomatous tissue response with asteroid body

The histologic variety and transformation in cutaneous cryptococcosis with acute lymphocytic leukemia before antifungal treatment and after the start of treatment were studied by the light and electron microscopic examinations. The initial cutaneous lesions before treatment revealed gelatinous tissue reactions, and Cryptococcus neoformans (serotype A) were isolated from the skin biopsy specimen and blood. However, later recurrent cutaneous lesions receiving antifungal treatment revealed suppurative granulomatous tissue reactions, and fungal cultures of the skin biopsy specimen changed to negative even though numerous yeasts stained with PAS were observed in skin lesions. Moreover, in the later lesion a few giant cells contained asteroid bodies without central spores. Ultrastructure of the later cutaneous lesions is presented.