Invasive rhino-orbital-cerebral aspergillosis in an immunocompetent patient

Leroy, Jordan; Vuotto, Fanny; Le, Vanbang; Cornu, Marjorie; François, Nadine; Marceau, L.; Fichet, C.; Loridant, Séverine; Sendid, Boualem

doi:10.1016/j.mycmed.2020.101002

Cited by 8 publications

(5 citation statements)

References 19 publications

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“…There were no obvious identified risk factors for the development of invasive fungal RS as the patient did not have immunodeficiency or disease such as diabetes mellitus [ 25 ] or haematological malignancy [ 18 ]. These findings are consistent with the previously reported cases of invasive rhino-orbital-cerebral aspergillosis and invasive oronasal aspergillosis in immunocompetent patients [ 19 , 21 , 22 , 23 ].…”

Section: Discussion and Literature Reviewsupporting

confidence: 93%

Sinonasal Orbital Apex Syndrome, Horner Syndrome and Pterygopalatine Fossa Infection: A Case Report and Mini-Review

Benites,

Urbančič,

Bardales

et al. 2023

Life

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This paper presents a literature review and a case of an 83-year-old otherwise healthy female patient with a history of recent syncope, a sudden-onset right-sided temporal headache, diplopia, and vision loss. An exam revealed right-sided upper eyelid ptosis, myosis, vision loss, ophthalmoplegia, and a positive relative afferent pupillary defect on the right eye. CT showed sphenoid sinus opacification, eroded lateral sinus wall, Vidian canal, disease extension to the posterior ethmoid air cells, orbital apex, medial orbital wall, and pterygopalatine fossa. An orbital apex syndrome (Jacod’s syndrome), Horner syndrome, and pterygopalatine fossa infection were diagnosed due to the acute invasive fungal sinusitis developed from a sphenoid sinus fungal ball. The patient was treated with antimicrobial therapy and transnasal endoscopic surgery twice to decompress the orbital apex, drain the abscess and obtain specimens for analysis. The right-sided ptosis, visual loss, ophthalmoplegia, and headache resolved entirely. No immune or comorbid diseases were identified, microbiological and histopathological analyses were negative, and MRI could not be performed on the presented patient. For that reason, the diagnostic procedure was non-standard. Nevertheless, the treatment outcome of this vision and life-threatening disease was satisfactory. Treating the fungal ball in an older or immunocompromised patient is essential to prevent invasive fungal rhinosinusitis and fatal complications.

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Section: Discussion and Literature Reviewsupporting

confidence: 93%

Sinonasal Orbital Apex Syndrome, Horner Syndrome and Pterygopalatine Fossa Infection: A Case Report and Mini-Review

Benites,

Urbančič,

Bardales

et al. 2023

Life

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“…Invasive aspergillosis, i.e., deep infections by an Aspergillus species, are among the most common opportunistic mold infections that can cause potentially life-threatening disease in those with chronic neutropenia, or inherited or acquired immunodeficiencies, as well as in those undergoing allogeneic hematopoietic stem cell transplant, solid organ transplant, or prolonged corticosteroid use (Li et al, 2008;Patterson et al, 2016). Among these, pulmonary infection and rhino-(facial)orbital-cerebral mycosis (ROCM) are the most problematic types (Patterson et al, 2016;Leroy et al, 2020;Hu et al, 2021). Second only to Aspergillus fumigatus for lung infections and ROCM (Alsalman et al, 2017;Rudramurthy and Paul, 2019;Hu et al, 2021), Aspergillus flavus is the most frequently encountered species in invasive sinusitis (Bakhshaee et al, 2016;Alsalman et al, 2017); in 9% of cases of ROCM, A. flavus is listed as the causal organism (Hu et al, 2021), while other species have remained unidentified in the literature (Hu et al, 2021).…”

Section: Introductionmentioning

confidence: 99%

Fatal Rhinofacial Mycosis Due to Aspergillus nomiae: Case Report and Review of Published Literature

Zhou

Houbraken

et al. 2020

Front. Microbiol.

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BackgroundA 73-year-old female suffering from acute myeloid leukemia presented with progressive rhinofacial mycosis. Suspecting it to be mucormycosis, the antifungal amphotericin B (AMB) was administered empirically, but the patient did not respond as planned. The fungus was then isolated from the biopsied tissue and morphologically identified as a species of Aspergillus. Necrosis progressed and she died of cerebral hemorrhage. Since Aspergillus flavus is susceptible to AMB, and several other Aspergillus species can be misidentified as A. flavus, the observed resistance necessitated a re-examination of the fungal isolate.MethodsThe fungal strain was re-isolated and re-examined morphologically. Additionally, genomic DNA was extracted from the fungus and sequences were obtained from three genomic regions [the rDNA internal transcribed spacer (ITS) region, and portions of the β-tubulin and calmodulin genes] to more accurately identify this Aspergillus strain. Its antifungal susceptibility was assessed using multiple compounds and our findings were compared with literature data.ResultsThe fungal culture again yielded an Aspergillus isolate morphologically identical to A. flavus. Molecular analyses, however, revealed the strain to be A. nomiae, a close relative of A. flavus in section Flavi, and it exhibited resistance to AMB. Reviewing the literature, only five other cases of A. nomiae infection in humans have been reported worldwide.Conclusion and Clinical ImportanceThe rhinofacial mycosis of the patient was actually due to A. nomiae. The initial misidentification of the fungus, coupled with its resistance to AMB, could be the reason treatment did not help the patient. We postulate that clinical A. nomiae infections may be underreported and that accurate and speedy pathogen identification is important so that an effective antifungal regimen can be administered.

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“…Aspergillus is known to cause pulmonary aspergillosis in COVID-19 patients[ 22 ] as well as rhino-orbito-cerebral syndrome in non-COVID-19. [ 23 ] To the best of our knowledge, there are no case reports of rhino-orbito-cerebral aspergillosis with COVID-19. The dual infection caused by mucor and aspergillus has also been described in non-COVID-19 patients,[ 24 ] but not in COVID-19.…”

Section: Discussionmentioning

confidence: 99%

Rhino-Orbito-Cerebral Mycosis and COVID-19

Kulkarni¹,

Pujari²,

Gupta³

et al. 2022

Annals of Indian Academy of Neurology

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Background: There has been an increase an alarming rise in invasive mycoses during COVID-19 pandemic, especially during the second wave. Aims: Compare the incidence of invasive mycoses in the last three years and study the risk factors, manifestations and outcomes of mycoses in the COVID era. Methodology: Multicentric study was conducted across 21 centres in a state of western India over 12-months. The clinico-radiological, laboratory and microbiological features, treatment and outcomes of patients were studied. We also analysed yearly incidence of rhino-orbito-cerebral mycosis. Results: There was more than five-times rise in the incidence of invasive mycoses compared to previous two-years. Of the 122 patients analysed, mucor, aspergillus and dual infection were seen in 86.9%, 4.1%, and 7.4% respectively. Fifty-nine percent had simultaneous mycosis and COVID-19 while rest had sequential infection. Common presenting features were headache (91%), facial pain (78.7%), diplopia (66.4%) and vison loss (56.6%). Rhino-orbito-sinusitis was present in 96.7%, meningitis in 6.6%, intracranial mass lesions in 15.6% and strokes in 14.8%. A total of 91.8% patients were diabetic, while 90.2% were treated with steroids during COVID-19 treatment. Mortality was 34.4%. Conclusion: Invasive fungal infections having high mortality and morbidity have increased burden on already overburdened healthcare system. Past illnesses, COVID-19 itself and its treatment and environmental factors seem responsible for the rise of fungal infection. Awareness and preventive strategies are the need of hours and larger studies are needed for better understanding of this deadly disease.

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Invasive rhino-orbital-cerebral aspergillosis in an immunocompetent patient

Cited by 8 publications

References 19 publications

Sinonasal Orbital Apex Syndrome, Horner Syndrome and Pterygopalatine Fossa Infection: A Case Report and Mini-Review

Sinonasal Orbital Apex Syndrome, Horner Syndrome and Pterygopalatine Fossa Infection: A Case Report and Mini-Review

Fatal Rhinofacial Mycosis Due to Aspergillus nomiae: Case Report and Review of Published Literature

Rhino-Orbito-Cerebral Mycosis and COVID-19

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