A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature review

Abstract Background Systemic 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) neonat...

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Main Authors: Alexandra Mpakosi, Maria Siopi, Maria Demetriou, Vasiliki Falaina, Martha Theodoraki, Joseph Meletiadis
Format: Article
Language:English
Published: BMC 2022-08-01
Series:BMC Pediatrics
Subjects:
Online Access:https://doi.org/10.1186/s12887-022-03518-5
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author Alexandra Mpakosi
Maria Siopi
Maria Demetriou
Vasiliki Falaina
Martha Theodoraki
Joseph Meletiadis
author_facet Alexandra Mpakosi
Maria Siopi
Maria Demetriou
Vasiliki Falaina
Martha Theodoraki
Joseph Meletiadis
author_sort Alexandra Mpakosi
collection DOAJ
description Abstract Background Systemic 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 presentation A 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. Conclusions Due 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.
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spelling doaj.art-3e03cdff96ac481298709f4c041bb4952022-12-22T03:59:11ZengBMCBMC Pediatrics1471-24312022-08-012211910.1186/s12887-022-03518-5A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature reviewAlexandra Mpakosi0Maria Siopi1Maria Demetriou2Vasiliki Falaina3Martha Theodoraki4Joseph Meletiadis5Department of Microbiology, General Hospital of Nikaia Agios PanteleimonClinical Microbiology Laboratory, Attikon University Hospital, Medical School, National and Kapodistrian University of AthensDepartment of Microbiology, Metaxa Memorial Anticancer Hospital of PiraeusNeonatal Intensive Care Unit, General Hospital of Nikaia Agios PanteleimonNeonatal Intensive Care Unit, General Hospital of Nikaia Agios PanteleimonClinical Microbiology Laboratory, Attikon University Hospital, Medical School, National and Kapodistrian University of AthensAbstract Background Systemic 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 presentation A 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. Conclusions Due 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.https://doi.org/10.1186/s12887-022-03518-5Exophiala dermatitidisPremature neonateExtremely low birth weightFungemiaBlack yeast-like fungus
spellingShingle Alexandra Mpakosi
Maria Siopi
Maria Demetriou
Vasiliki Falaina
Martha Theodoraki
Joseph Meletiadis
A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature review
BMC Pediatrics
Exophiala dermatitidis
Premature neonate
Extremely low birth weight
Fungemia
Black yeast-like fungus
title A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature review
title_full A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature review
title_fullStr A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature review
title_full_unstemmed A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature review
title_short A fatal neonatal case of fungemia due to Exophiala dermatitidis—case report and literature review
title_sort fatal neonatal case of fungemia due to exophiala dermatitidis case report and literature review
topic Exophiala dermatitidis
Premature neonate
Extremely low birth weight
Fungemia
Black yeast-like fungus
url https://doi.org/10.1186/s12887-022-03518-5
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