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1.
J Pediatr Hematol Oncol ; 42(3): e193-e194, 2020 04.
Artigo em Inglês | MEDLINE | ID: mdl-30676435

RESUMO

Trichosporon asahii is a rare opportunistic fungal pathogen that causes fatal systemic infection in immunocompromised patients. Neutropenia developing due to malignancies is an important risk factor for fungal infection. Invasive infections due to T. asahii can be divided into disseminated and localized forms. The disseminated form is more common and usually occurs in neutropenic patients. The patient typically has an acute febrile illness that progresses rapidly to multiorgan failure. Here, we are presenting a case of fungal sepsis by invasive T. asahii in a 1-year-old child with Wilms Tumor. To the best of our knowledge, this is the first time that fungal sepsis due to T. asahii has been reported in a Wilms tumor patient. The incidence of rare invasive fungal infections is increasing in immunocompromised patients in whom management becomes difficult due to their heterogenous antifungal susceptibility pattern and intrinsic resistance to the standard antifungal agents that are routinely given. The patient was admitted with high spiking fever, and his laboratory investigations suggested neutropenia. T. asahii was isolated from the blood culture, for which he was started on inj. voriconozole. After 14 days of treatment, the fungus was cleared out from the patient's blood.


Assuntos
Infecções Relacionadas a Cateter/microbiologia , Cateteres de Demora/efeitos adversos , Fungemia/imunologia , Hospedeiro Imunocomprometido , Tricosporonose/imunologia , Antifúngicos/uso terapêutico , Protocolos de Quimioterapia Combinada Antineoplásica , Dactinomicina , Humanos , Lactente , Neoplasias Renais/tratamento farmacológico , Masculino , Tricosporonose/tratamento farmacológico , Vincristina , Voriconazol/uso terapêutico
2.
BMJ Case Rep ; 12(6)2019 Jun 18.
Artigo em Inglês | MEDLINE | ID: mdl-31217208

RESUMO

Association between pulmonary disease and IgA nephropathy (IgAN) has been previously reported. However, no association has been reported between hypersensitivity pneumonitis (HP) and IgAN. Here, we report about a patient with no particular medical history, who experienced worsening dyspnoea in the course of 1 month, with ground-glass opacity on chest CT and no improvement after antibiotic therapy. The patient was diagnosed as having HP based on the history of antigen exposure, detection of Trichosporon asahii-specific antibodies and bronchoscopy findings. Concomitantly, findings of renal biopsy revealed the IgAN diagnosis. The patient underwent corticosteroid therapy, with good outcomes for both HP and IgAN. This is the first report in the literature to describe summer-type HP complicated with IgAN.


Assuntos
Corticosteroides/uso terapêutico , Ar Condicionado/efeitos adversos , Alveolite Alérgica Extrínseca/microbiologia , Dispneia/microbiologia , Glomerulonefrite por IGA/microbiologia , Tricosporonose/diagnóstico , Alveolite Alérgica Extrínseca/tratamento farmacológico , Alveolite Alérgica Extrínseca/imunologia , Anticorpos Antifúngicos , Broncoscopia , Tosse , Dispneia/etiologia , Glomerulonefrite por IGA/tratamento farmacológico , Glomerulonefrite por IGA/imunologia , Habitação , Humanos , Imunoglobulina A/imunologia , Masculino , Pessoa de Meia-Idade , Radiografia Torácica , Estações do Ano , Resultado do Tratamento , Tricosporonose/tratamento farmacológico , Tricosporonose/imunologia , Tricosporonose/fisiopatologia
3.
Br J Dermatol ; 180(5): 1221-1225, 2019 05.
Artigo em Inglês | MEDLINE | ID: mdl-30117151

RESUMO

Deficiency of CARD9 (caspase recruitment domain-containing protein 9) has been reported in individuals with recurrent and invasive fungal infections. We report on a patient who first had Trichosporon asahii affecting the skin then Candida albicans infections involving the digestive tract and knee joint, along with elevated serum IgE. After stimulation with C. albicans, peripheral blood mononuclear cells of this patient produced less tumour necrosis factor-α, interferon-γ and interleukin-17 than those of healthy controls. Furthermore, the serum IgE levels of this patient were positively correlated with the severity of fungal infection during the course of treatment. Sanger sequencing identified one homozygous frameshift mutation (p.D274fsX60) in CARD9. We further performed a review including 48 cases with CARD9 deficiency. According to the data published previously, CARD9-deficient patients demonstrated obviously elevated IgE in serum (median 1300 IU mL-1 ), which could distinguish them from otherwise healthy people with fungal infections (area under the curve 0·94, P < 0·001). Patients carrying the mutations Q289X and Q295X had a higher mortality rate (24% vs. 0%, P < 0·05). Patients with the mutations R18W, R35Q, R70W, G72S or Y91H in the CARD domain, and the nonsense mutation Q295X in the coiled-coil domain, seemed to be more prone to Candida infections (90% vs. 20%, P < 0·005) and central nervous system infections (60% vs. 12%, P < 0·005).


Assuntos
Proteínas Adaptadoras de Sinalização CARD/genética , Candidíase Mucocutânea Crônica/diagnóstico , Predisposição Genética para Doença , Tricosporonose/diagnóstico , Candida albicans/imunologia , Candida albicans/isolamento & purificação , Candidíase Mucocutânea Crônica/genética , Candidíase Mucocutânea Crônica/imunologia , Candidíase Mucocutânea Crônica/microbiologia , Análise Mutacional de DNA , Mutação da Fase de Leitura , Humanos , Masculino , Recidiva , Pele/microbiologia , Trichosporon/imunologia , Trichosporon/isolamento & purificação , Tricosporonose/genética , Tricosporonose/imunologia , Tricosporonose/microbiologia , Adulto Jovem
4.
BMJ Case Rep ; 20182018 Jan 06.
Artigo em Inglês | MEDLINE | ID: mdl-29306852

RESUMO

A 59-year-old man with a history of peripheral vascular disease status post femoral popliteal bypass presented with critical limb ischaemia of the left leg. An arterial Doppler ultrasound showed an occluded graft requiring an above knee amputation. Five days after surgery, the patient developed fever, leucocytosis, significant stump swelling and pain, and serosanguinous discharge from his wound. Wound swab cultures from the stump grew Trichosporon asahii A venous Doppler ultrasound revealed extensive thrombosis of the left lower extremity. Biopsy of the left thigh muscle showed necrotic thrombus with fungal hyphae in the clotted blood vessel. The left femoral vein was subsequently resected, and the excised venous tissue also grew T. asahii The patient was successfully treated with voriconazole based on antifungal susceptibilities. This case describes an invasive fungal infection in the absence of typical immunosuppressive conditions commonly associated with Trichosporon spp. It also illustrates the role of a combination of antimicrobial and surgical management in achieving cure.


Assuntos
Amputação Cirúrgica/efeitos adversos , Hospedeiro Imunocomprometido/imunologia , Complicações Pós-Operatórias/imunologia , Tromboflebite/imunologia , Tricosporonose/imunologia , Amputação Cirúrgica/métodos , Veia Femoral/microbiologia , Veia Femoral/cirurgia , Humanos , Extremidade Inferior/irrigação sanguínea , Extremidade Inferior/cirurgia , Masculino , Pessoa de Meia-Idade , Complicações Pós-Operatórias/microbiologia , Tromboflebite/microbiologia , Trichosporon , Tricosporonose/microbiologia
5.
J Pediatr Hematol Oncol ; 40(2): 156-158, 2018 03.
Artigo em Inglês | MEDLINE | ID: mdl-28562512

RESUMO

A 4-year-old girl with severe aplastic anemia and 2 previous failed T-depleted haploidentical peripheral blood stem cell transplants developed persistent neutropenic fever and multiple erythematous maculopapular rashes 2 days after her third T-replete haploidentical bone marrow transplant. Skin biopsy confirmed the diagnosis of Trichosporon asahii infection. She was on caspofungin prophylaxis which is not effective against Trichosporon. A high index of suspicion, prompt investigation, and appropriate treatment with voriconazole for 4 months was instrumental in controlling the infection and she remains well presently 9 months posttransplant with full donor chimerism and free from infection.


Assuntos
Anemia Aplástica/complicações , Exantema/imunologia , Febre/imunologia , Hospedeiro Imunocomprometido , Tricosporonose/imunologia , Anemia Aplástica/terapia , Antifúngicos/uso terapêutico , Pré-Escolar , Exantema/microbiologia , Feminino , Febre/microbiologia , Transplante de Células-Tronco Hematopoéticas , Humanos , Tricosporonose/tratamento farmacológico , Voriconazol/uso terapêutico
6.
Mycopathologia ; 179(1-2): 11-20, 2015 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-25179349

RESUMO

Trichosporon asahii is the major cause of invasive trichosporonosis, but little is known about the host immune response to this pathogen. In this study, the early transcriptional response of human monocyte-like THP-1 cells to T. asahii infection was evaluated using cDNA microarray and 1,315 differentially expressed genes were identified. The up-regulated genes were mostly involved in both innate and adaptive immune responses, as well as apoptosis and anti-apoptosis processes. Genes encoding the pro-inflammatory cytokines TNF-α, IL-1ß, IL18 and IL-23α, along with the both C-C motif and C-X-C motif chemokines were strongly up-regulated, suggesting that THP-1 cells can mount a powerful inflammatory response to T. asahii infection. Genes encoding pattern recognition receptors were found up-regulated, such as dendritic cell-specific intercellular adhesion molecule 3-grabbing nonintegrin, cluster of differentiation 36 and the long pentraxin 3. Genes encoding members of the dual-spasticity phosphates family were up-regulated, and these genes were considered as a negative feedback mechanism to prevent excessive inflammatory response. The down-regulated genes in T. asahii-infected THP-1 cells were predominantly associated with cell cycle, mitosis, cell division and DNA repair. Thus, our study defines the early transcriptional response of monocyte-like THP-1 cells to T. asahii infection and provides a foundation for further investigations into the pathogenesis of T. asahii infection.


Assuntos
Interações Hospedeiro-Patógeno/imunologia , Monócitos/imunologia , Trichosporon/imunologia , Tricosporonose/imunologia , Antígenos CD/biossíntese , Proteína C-Reativa/biossíntese , Antígenos CD36/biossíntese , Moléculas de Adesão Celular/biossíntese , Linhagem Celular Tumoral , DNA Fúngico/genética , Interações Hospedeiro-Patógeno/genética , Humanos , Interleucina-18/biossíntese , Interleucina-1beta/biossíntese , Subunidade p19 da Interleucina-23/biossíntese , Análise em Microsséries , Mitose/genética , Componente Amiloide P Sérico/biossíntese , Transcrição Gênica/genética , Tricosporonose/microbiologia , Fator de Necrose Tumoral alfa/biossíntese
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