Multiple Brain Abscesses Caused by Trichosporon inkin in a Patient with X-Linked Chronic Granulomatous Disease (CGD) Successfully Treated with Antifungal Therapy

Hajjar, Joud; Restrepo, Alejandro; Javeri, Heta; Wiederhold, Nathan P.; Papanastassiou, Alexander; Patterson, Thomas F.

doi:10.1007/s10875-017-0419-1

Cited by 8 publications

(4 citation statements)

References 37 publications

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“…Whether some of these cases may be iatrogenic (e.g., vascular-catheter-associated) is not clear, although in some reports, this does not seem to be the case. In distinction to IC, CGD is associated with an increased risk of systemic disease via infection with another yeast, Trichosporon [ 93 , 94 , 95 , 96 , 97 ]. Given the challenges in the antifungal management of trichosporonosis, biopsies for pan-microbiological culture should be performed.…”

Section: Candidiasismentioning

confidence: 99%

Of Mycelium and Men: Inherent Human Susceptibility to Fungal Diseases

Vinh

2023

Pathogens

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In medical mycology, the main context of disease is iatrogenic-based disease. However, historically, and occasionally, even today, fungal diseases affect humans with no obvious risk factors, sometimes in a spectacular fashion. The field of “inborn errors of immunity” (IEI) has deduced at least some of these previously enigmatic cases; accordingly, the discovery of single-gene disorders with penetrant clinical effects and their immunologic dissection have provided a framework with which to understand some of the key pathways mediating human susceptibility to mycoses. By extension, they have also enabled the identification of naturally occurring auto-antibodies to cytokines that phenocopy such susceptibility. This review provides a comprehensive update of IEI and autoantibodies that inherently predispose humans to various fungal diseases.

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Section: Candidiasismentioning

confidence: 99%

Of Mycelium and Men: Inherent Human Susceptibility to Fungal Diseases

Vinh

2023

Pathogens

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“…Both Streptococcus intermedius and methicillin-susceptible Staphylococcus aureus emerged as causative organisms for liver abscess in one patient each in our cohort. A rare aggressive fungal organism that was detected in our cohort was Trichosporon species, previously reported in six patients with CGD, four of whom presented with pneumonia (26-28), while one patient developed lung abscesses (29) and one patient was diagnosed with multiple brain abscesses (30). Most patients required surgical resection despite antifungal therapy.…”

Section: Discussionmentioning

confidence: 56%

Chronic granulomatous disease in the United Arab Emirates: clinical and molecular characteristics in a single center

Al Kuwaiti,

Al Dhaheri,

Al Hassani

et al. 2023

Front. Immunol.

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BackgroundChronic granulomatous disease (CGD) is a genetic disorder caused by defective oxidative burst within phagocytes, manifesting as recurrent, severe infections as well as hyperinflammation.ObjectiveThis is the first report from the United Arab Emirates (UAE) to describe the demographic, clinical, laboratory, radiological, and genetic characteristics of patients with CGD.MethodsThis is a retrospective study that was conducted at Tawam Hospital in the UAE on patients with confirmed CGD between 2017 and 2022.ResultsA total of 14 patients were diagnosed with CGD, of whom 13 patients had autosomal recessive (AR) CGD due to NCF1 deficiency. Consanguinity was noted in all patients with AR CGD, whereas positive family history was identified in 50% of cases. The median age of onset of symptoms was 24 months, while the median age at diagnosis was 72 months. Lymphadenitis was the most common clinical feature identified in 71% of patients. Other common infectious manifestations included abscess formation (57%), pneumonia (50%), invasive aspergillosis (21%), oral thrush (14%), and sepsis (14%). Disseminated trichosporonosis was reported in one patient. Autoimmune and inflammatory manifestations included celiac disease in two patients, diabetes mellitus and asymptomatic colitis in one patient each. Genetic analysis was performed in all patients; NCF1 deficiency was diagnosed in 13 (93%) patients, with c.579G>A being the most prevalent pathogenic variant identified. The treatment modalities, as well as treatment of acute infections, treatment modalities included antimicrobial prophylaxis in 12 (86%) patients and hematopoietic stem cell transplant in six patients (42%).ConclusionThis is the first report from the UAE describing the clinical and molecular characteristics of patients with CGD. The homozygous variant c.579G>A causing NCF1 deficiency can be considered as a founder mutation for AR CGD in the UAE.

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“…Although there have been reports of abscesses and meningitis, fungal aneurysms caused by trichosporonosis have not been reported. [1,5,6,8,[10][11][12][13][14][15] is is the first report of an intracranial fungal aneurysm caused by trichosporonosis. Trichosporon species are naturally resistant to candin antifungals, so there is a risk of breakthrough infection in case of inadvertent use.…”

Section: Discussionmentioning

confidence: 99%

A case of subarachnoid and intracerebral hemorrhages complicated by trichosporonosis

Shibao

Kaburaki

Saito

et al. 2022

Surgical Neurology International

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Background: Trichosporonosis has an extremely poor prognosis. In this report, we describe a case of subarachnoid hemorrhage and intracerebral hemorrhage due to a fungal aneurysm caused by Trichosporon. Case Description: A 71-year-old woman who experienced subcortical hemorrhage developed a subarachnoid hemorrhage. Endovascular parent artery occlusion was performed for a fungal aneurysm in the left posterior cerebral artery caused by Trichosporon. After surgery, voriconazole and liposomal amphotericin B were administered. The patient died of massive left putamen hemorrhage. Conclusion: Effective treatment for intracranial hemorrhage due to trichosporonosis has not yet been established and an accumulation of cases is required.

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Multiple Brain Abscesses Caused by Trichosporon inkin in a Patient with X-Linked Chronic Granulomatous Disease (CGD) Successfully Treated with Antifungal Therapy

Cited by 8 publications

References 37 publications

Of Mycelium and Men: Inherent Human Susceptibility to Fungal Diseases

Of Mycelium and Men: Inherent Human Susceptibility to Fungal Diseases

Chronic granulomatous disease in the United Arab Emirates: clinical and molecular characteristics in a single center

A case of subarachnoid and intracerebral hemorrhages complicated by trichosporonosis

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