Disseminated Fusariosis Secondary to Neuroblastoma with Fatal Outcome

Silva, Gleiciere Maia; Silveira, Alice Rangel Cristiane; Betânia, Claudia Abreu Rodrigues; Macêdo, Danielle Patrícia Cerqueira; Neves, Rejane Pereira

doi:10.1007/s11046-013-9674-8

Cited by 8 publications

(6 citation statements)

References 20 publications

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“…Most Fusarium species have low susceptibilities to the majority of azole antifungals (Tupaki-Sreepurna et al, 2017;Rotjanapan et al, 2018;Herkert et al, 2019) and patients with IFs have high mortality rates (Esnakula et al, 2013;Silva et al, 2013;Okada et al, 2018). Voriconazole (VRC) and amphotericin B (AMB) are the recommended treatment options for localised infections and IFs (Efeİris et al, 2016;Okada et al, 2018).…”

Section: Introductionmentioning

confidence: 99%

A 23 bp cyp51A Promoter Deletion Associated With Voriconazole Resistance in Clinical and Environmental Isolates of Neocosmospora keratoplastica

et al. 2020

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In the fungal pathogen Aspergillus fumigatus, resistance to azole antifungals is often linked to mutations in CYP51A, a gene that encodes the azole antifungal drug target lanosterol 14α-demethylase. The aim of this study was to investigate whether similar changes could be associated with azole resistance in a Malaysian Fusarium solani species complex (FSSC) isolate collection. Most (11 of 15) clinical FSSC isolates were Neocosmospora keratoplastica and the majority (6 of 10) of environmental isolates were Neocosmospora suttoniana strains. All 25 FSSC isolates had high minimum inhibitory concentrations (MICs) for itraconazole and posaconazole, low MICs for amphotericin B, and various (1 to >32 mg/l) voriconazole susceptibilities. There was a tight association between a 23 bp CYP51A promoter deletion and high (>32 mg/l) voriconazole MICs; of 19 FSSC strains sequenced, nine isolates had voriconazole MICs > 32 mg/l, and they all contained the 23 bp CYP51A promoter deletion, although it was absent in the ten remaining isolates with low (≤12 mg/l) voriconazole MICs. Surprisingly, this association between voriconazole resistance and the 23 bp CYP51A promoter deletion held true across species boundaries. It was randomly distributed within and across species boundaries and both types of FSSC isolates were found among environmental and clinical isolates. Three randomly selected N. keratoplastica isolates with low (≤8 mg/l) voriconazole MICs had significantly lower (1.3-7.5 times) CYP51A mRNA expression levels than three randomly selected N. keratoplastica isolates with high (>32 mg/l) voriconazole MICs. CYP51A expression levels, however, were equally strongly induced (∼6,500-fold) by voriconazole in two representative strains reaching levels, after 80 min of induction, that were comparable to those of CYP51B. Our results suggest that FSSC isolates with high voriconazole MICs have a 23 bp CYP51A promoter deletion that provides a potentially useful marker for voriconazole resistance in FSSC isolates. Early detection of possible voriconazole resistance is critical for choosing the correct treatment option for patients with invasive fusariosis.

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Section: Introductionmentioning

confidence: 99%

A 23 bp cyp51A Promoter Deletion Associated With Voriconazole Resistance in Clinical and Environmental Isolates of Neocosmospora keratoplastica

et al. 2020

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“…The current treatment for invasive Fusarium infection is antifungal therapy with immunotherapy and surgical debridement, if necessary . Due to the rarity of infection and growing antifungal resistance, however, there is no consensus on optimal treatment with regards to the most effective antifungal, optimum dosing, value of antifungal combination versus monotherapy, and the extent of adjunctive surgery . Some reports cite amphotericin B as the antifungal treatment of choice although it has significant side effects, including nephrotoxicity .…”

Section: Discussionmentioning

confidence: 99%

“…Due to the rarity of infection and growing antifungal resistance, however, there is no consensus on optimal treatment with regards to the most effective antifungal, optimum dosing, value of antifungal combination versus monotherapy, and the extent of adjunctive surgery . Some reports cite amphotericin B as the antifungal treatment of choice although it has significant side effects, including nephrotoxicity . Voriconazole has demonstrated promising results in the treatment of fusariosis .…”

Section: Discussionmentioning

confidence: 99%

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Monoarthritis in a 28‐Year‐Old Man With Juvenile Idiopathic Arthritis

Curry

Matzkin

et al. 2015

Arthritis Care & Research

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A 28-year-old white male presented to his outpatient rheumatologist with monoarticular left knee pain, erythema, warmth, significant swelling, and immobility due to pain. He had a history of recurrent episodes of refractory inflammatory arthritis mostly affecting the knees. The current episode began about 1 week prior to presentation and consisted of swelling in the left knee with difficulty bearing weight and ambulating. Similar symptoms had recurred for the previous 3 months. No other joints were painful. He denied fever or chills. He also noted a diffuse maculopapular and pustular rash over his back, chest, hands, and trunk that started about 2 weeks prior and was painful and pruritic. He had some slight chronic bilateral eye pain near his baseline without associated visual loss and slight photophobia.His recent history was significant for recurrent inflammatory arthritis in the right (contralateral) knee with large effusions refractory to medical therapy requiring 2 synovectomies. The most recent synovectomy on the right knee was performed 1 year prior, and this successfully controlled his effusion and pain in this knee. He had been maintained on methotrexate 25 mg subcutaneously weekly, but had several flares in the left knee in the 3 months prior to the current presentation requiring high-dose systemic glucocorticoids and intraarticular corticosteroids. Multiple prior therapies were ineffective or not tolerated (detailed below in Past medical history). Recurrent episodes in the left knee consisted of large effusions, reduced range of motion, and inability to bear weight. In these 3 months, he had a total of 6 aspirations (synovial white blood cell counts ranging from 17.2 to 98.2 x 10 3 cells/mm 3 ) with multiple corticosteroid injections of the left knee, and none of which revealed crystals on microscopy or organisms on Gram stain or culture.The patient was hospitalized about 3 weeks prior to the current presentation for left knee pain. During this hospitalization, 70 ml of cloudy synovial fluid were aspirated. Synovial fluid analysis at that time showed 17.3 x 10 3 white blood cells/mm 3 (normal 4.4-11 x 10 3 cells/mm 3 ), 74% neutrophils, 21% monocytes, 5% lymphocytes, no crystals, and no organisms on Gram stain. Cultures were without growth. At that time, triamcinolone 80 mg with lidocaine was injected into the left knee. He was discharged and a few days later received 1 dose of methylprednisolone 1 gram intravenously and infliximab infusion at 3 mg/kg as an outpatient. Past medical historyThe patient's medical history was notable for history of uveitis and iritis since the age of 2 years and reported polyarticular juvenile idiopathic arthritis (JIA) since the age of 5 years, mostly involving both knees. He was negative for antinuclear antibody, rheumatoid factor (RF), HLA-B27, and anti-cyclic citrullinated peptide antibodies. The inflammatory arthritis in his knees was refractory to many diseasemodifying antirheumatic drugs (DMARDs), biologic DMARDs, and other systemic immunosuppressants, including gl...

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“…Amphotericin B seems to be the only antifungal to which most FSSC species are susceptible [ 29 , 30 ]. This hinders the efficacy of treating immunocompromised patients with IF [ 33 , 34 , 35 , 36 ]. The widespread use of fungicides in agriculture [ 37 ] has led to the rise and global spread of azole resistant Aspergillus fumigatus isolates [ 38 ] into hospital settings [ 39 , 40 , 41 ], some of which are beginning to exhibit resistance to multiple classes of antifungals [ 42 ].…”

Section: Introductionmentioning

confidence: 99%

Morphology, Phenotype, and Molecular Identification of Clinical and Environmental Fusarium solani Species Complex Isolates from Malaysia

James

Santhanam

Zakaria

et al. 2022

JoF

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Fusarium infections in humans (fusariosis) and in economically important plants involve species of several Fusarium species complexes. Species of the Fusarium solani species complex (FSSC) are the most frequent cause of human fusariosis. The FSSC comprises more than 60 closely related species that can be separated into three major clades by multi-locus sequence typing (MLST) using translation elongation factor 1-alpha (TEF1-α) and RNA polymerase II (RPB2) DNA sequences. The MLST nomenclature for clade 3 of the FSSC assigns numbers to species types (e.g., FSSC 2) and lowercase letters to identify unique haplotypes. The aim of this study was to analyse the genotypic and phenotypic characteristics of 15 environmental and 15 clinical FSSC isolates from Malaysia. MLST was used for the genotypic characterisation of FSSC isolates from various locations within Malaysia, which was complemented by their morphological characterisation on potato dextrose and carnation leaf agar. MLST identified eight different FSSC species: thirteen Fusarium keratoplasticum (i.e., FSSC 2), six Fusarium suttonianum (FSSC 20), five Fusarium falciforme (FSSC 3+4), two Fusarium cyanescens (FSSC 27), and one each of Fusarium petroliphilum (FSSC 1), Fusarium waltergamsii (FSSC 7), Fusarium sp. (FSSC 12), and Fusarium striatum (FSSC 21). Consistent with previous reports from Malaysia, most (11 of 15) clinical FSSC isolates were F. keratoplasticum and the majority (9 of 15) of environmental isolates were F. suttonianum (5) or F. falciforme (4) strains. The taxonomic relationships of the isolates were resolved phylogenetically. The eight Fusarium species also showed distinct morphological characteristics, but these were less clearly defined and reached across species boundaries. Although TEF1-α and RPB2 sequences were sufficient for the species identification of most FSSC isolates, a more precise MLST scheme needs to be established to reliably assign individual isolates of the species-rich FSSC to their geographically-, epidemiologically-, and host-associated sub-lineages.

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Disseminated Fusariosis Secondary to Neuroblastoma with Fatal Outcome

Cited by 8 publications

References 20 publications

A 23 bp cyp51A Promoter Deletion Associated With Voriconazole Resistance in Clinical and Environmental Isolates of Neocosmospora keratoplastica

A 23 bp cyp51A Promoter Deletion Associated With Voriconazole Resistance in Clinical and Environmental Isolates of Neocosmospora keratoplastica

Monoarthritis in a 28‐Year‐Old Man With Juvenile Idiopathic Arthritis

Morphology, Phenotype, and Molecular Identification of Clinical and Environmental Fusarium solani Species Complex Isolates from Malaysia

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