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Lodderomyces elongisporus fungemia in a patient with previous cardiac surgery: Case report and review of literature.

ABSTRACT: Lodderomyces elongisporus is a rare cause of invasive fungal infections. Most phenotypic tests that are routinely used for identification of yeasts fail to identify this organism. However, chromogenic media for yeasts, MALDI-TOF MS and DNA sequencing can be used for correct identification. We report a case of fungemia complicated by infective endocarditis and intracerebral bleeding in a pediatric patient with previous cardiac surgery.

SUBMITTER: Gourav S

PROVIDER: S-EPMC10240516 | biostudies-literature | 2023 Jun

REPOSITORIES: biostudies-literature

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<i>Lodderomyces elongisporus</i> fungemia in a patient with previous cardiac surgery: Case report and review of literature.

Gourav Sudesh S Xess Immaculata I Xess Ashit Bhushan AB Yadav Renu Kumari RK Ramakrishnan Sivasubramanian S Singh Gagandeep G

Medical mycology case reports 20230405

<i>Lodderomyces elongisporus</i> is a rare cause of invasive fungal infections. Most phenotypic tests that are routinely used for identification of yeasts fail to identify this organism. However, chromogenic media for yeasts, MALDI-TOF MS and DNA sequencing can be used for correct identification. We report a case of fungemia complicated by infective endocarditis and intracerebral bleeding in a pediatric patient with previous cardiac surgery. ...[more]

PMID: 37283720

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Project description:Lodderomyces elongisporus is a recently emerging yeast pathogen predominantly reported in adult patients who had immunosuppression and/or intravenous access devices. Here, we report a fungemia outbreak caused by L. elongisporus in a neonatal intensive care unit (NICU) in Delhi, India, from September 2021 to February 2022. All 10 neonates had low birthweight, and nine of the patients survived after amphotericin B treatment. Whole-genome sequence analyses of the patient isolates as well as those from other sources in India grouped them into two clusters: one cluster consists of isolates exclusively from stored apples and the other cluster includes isolates from patients, clinical environments, and stored apples. All outbreak strains from patients were closely related to each other and showed highly similar heterozygosity patterns across all 11 major scaffolds. While overall very similar, strains from the inanimate environment of the same neonatal intensive care unit showed loss of heterozygosity at scaffold 2 (NW_001813676) compared to the patient strains. Interestingly, evidence for recombination was found in all samples. All clinical strains were susceptible to 10 tested antifungal drugs, and comparisons with strains with high fluconazole MICs derived from the surface of stored apples revealed significant genome divergence between the clinical and apple surface strains, including 119 nonsynonymous single nucleotide polymorphisms (SNPs) in 24 triazole resistance-related genes previously found in other Candida spp. Together, our results indicate significant diversity, recombination, and persistence in the hospital setting and a high rate of evolution in this emerging yeast pathogen. IMPORTANCE Lodderomyces elongisporus was initially considered a teleomorph of Candida parapsilosis. However, DNA sequence analyses revealed it as a distinctive species. Invasive infections due to L. elongisporus have been reported globally. We report an outbreak of fungemia due to L. elongisporus in a NICU affecting 10 preterm, low-birthweight neonates during a period of 6 months. The outbreak investigation identified two environmental sites, the railing and the temperature panel of the neonate open care warmer, harboring L. elongisporus. Whole-genome sequencing confirmed that the neonate isolates were closely related to each other whereas strains from the inanimate clinical environment were related to clinical strains but showed a marked loss of heterozygosity. Further, L. elongisporus strains recovered previously from the surface of stored apples showed high fluconazole MICs and alterations in triazole resistance-related genes. Genome-wide SNP comparisons revealed recombination as an important source for genomic diversity during adaptation of L. elongisporus to different environments.

Project description:BackgroundSystemic infections caused by the black yeast-like fungus Exophiala dermatitidis are rare, but are associated with high mortality especially in immunocompromised patients. We report the first case of E. dermatitidis fungemia in a premature extremely low birth weight (ELBW) neonate who succumbed despite antifungal therapy with liposomal amphotericin (AMB) and fluconazole. A systematic review of all fungemia cases due to E. dermatitidis was also conducted aiming for a better understanding of the risk factors, treatment strategies and outcomes.Case presentationA male, ELBW premature neonate, soon after his birth, developed bradycardia, apnoea and ultimately necrotizing enterocolitis with intestinal perforation requiring surgical intervention. Meanwhile, he had also multiple risk factors for developing bloodstream infection, such as intubation, mechanical ventilation, central venous catheter (CVC), parenteral nutrition, empirical and prolonged antibiotic use. His blood cultures were positive, firstly for Acinetobacter junii and then for Klebsiella pneumoniae together with E. dermatitidis while on fluconazole prophylaxis and antibiotic empiric therapy. Despite the treatment with broad spectrum antibiotics, liposomal AMB and fluconazole, the newborn succumbed. A literature review identified another 12 E. dermatitidis bloodstream infections, mainly in patients with hematologic malignancies and solid organ transplant recipients (61%), with overall mortality 38% despite CVC removal and antifungal therapy.ConclusionsDue to the rarity of E. dermatitidis infections, little is known about the characteristics of this yeast, the identification methods and the optimal therapy. Identification by common biochemical tests was problematic requiring molecular identification. Resolution of neonatal fungemia is difficult despite proper antifungal therapy especially in cases with multiple and severe risk factors like the present one. Therapeutic intervention may include CVC removal and treatment for at least 3 weeks with an azole (itraconazole or fluconazole after susceptibility testing) or AMB monotherapy but not echinocandins or AMB plus azole combination therapy.

Dataset Information

Lodderomyces elongisporus fungemia in a patient with previous cardiac surgery: Case report and review of literature.

Publications

<i>Lodderomyces elongisporus</i> fungemia in a patient with previous cardiac surgery: Case report and review of literature.

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