Rhino-orbito-cerebral fungal infection associated with mixed Cladosporium halotolerans and Penicillium corylophilum, complicated by internal carotid artery occlusion and ischemic stroke: a case report.
Hidaka M, Kiyohara T, et al. • Journal of infection and chemotherapy : official journal of the Japan Society of Chemotherapy • 2026
A case of rhino-orbito-cerebral fungal infection caused by mixed Cladosporium halotolerans and Penicillium corylophilum was diagnosed after nearly 4 months by culturing a large volume (10 mL) of cerebrospinal fluid, with treatment by liposomal amphotericin B followed by voriconazole resulting in gradual clinical and radiologic improvement.
Key Findings
Background
Rhino-orbito-cerebral fungal infection can occur in patients without overt immunosuppression such as corticosteroid use or chemotherapy.
The patient was a 71-year-old man with no documented immunocompromising conditions
The infection presented with visual loss in the left eye
The case challenges the assumption that rhino-orbito-cerebral fungal infections occur only in immunocompromised patients
Results
A mixed fungal infection with Cladosporium halotolerans and Penicillium corylophilum was identified as the causative organisms.
Identification was achieved using Internal Transcribed Spacer rDNA sequencing and β-tubulin gene sequencing
This represents a rare mixed fungal infection involving two uncommon pathogens
Prior extensive investigations including surgical biopsy had failed to establish a definitive diagnosis
Results
Culturing a large volume (10 mL) of cerebrospinal fluid was key to achieving a definitive fungal diagnosis after nearly 4 months of diagnostic uncertainty.
The diagnosis could not be established for nearly 4 months despite extensive investigations including surgical biopsy
Fungal infection was eventually confirmed by culturing a large volume of 10 mL of CSF
The authors suggest that submitting an adequate volume of CSF may improve the diagnostic yield for identifying causative fungi
Results
The fungal infection was complicated by internal carotid artery occlusion and ischemic stroke.
Internal carotid artery occlusion was found in association with the rhino-orbito-cerebral infection
The occlusion was complicated by ischemic stroke
The authors suggest clinicians should consider fungal infection in patients with unexplained carotid artery occlusion and stroke
Results
Treatment with liposomal amphotericin B followed by voriconazole resulted in gradual clinical and radiologic improvement.
The antifungal regimen consisted of sequential therapy: liposomal amphotericin B first, followed by voriconazole
Improvement was characterized as 'gradual' in both clinical and radiologic terms
This treatment approach was used for a mixed infection involving Cladosporium halotolerans and Penicillium corylophilum
What This Means
This case report describes a 71-year-old man who developed a serious fungal infection spreading through his sinuses, eye socket, and brain — a condition called rhino-orbito-cerebral infection. What made this case unusual is that the patient had no obvious weakened immune system (such as from cancer treatment or steroid medications), which is the typical risk factor for this type of infection. The infection caused a blockage of a major artery supplying the brain (the internal carotid artery), which led to a stroke, and the patient lost vision in his left eye.
Despite extensive testing over nearly four months — including surgical tissue biopsies — doctors were unable to identify the cause until they cultured a larger-than-usual volume (10 mL) of cerebrospinal fluid (the fluid surrounding the brain and spinal cord). This test revealed not one but two rare fungal species: Cladosporium halotolerans and Penicillium corylophilum, identified through advanced genetic sequencing techniques. Once diagnosed, the patient was treated with antifungal medications (liposomal amphotericin B, then voriconazole), and his condition gradually improved both clinically and on imaging scans.
This research suggests that doctors should consider fungal infection as a possible cause when a patient has an unexplained blockage of the carotid artery and stroke, even if the patient does not appear to have a weakened immune system. It also highlights that using a larger volume of cerebrospinal fluid for fungal cultures may significantly improve the chances of identifying the responsible organism, potentially shortening the time to diagnosis in similar difficult cases.
Hidaka M, Kiyohara T, Yazaki M, Nakamura K, Wakisaka Y, Ishikawa K, et al.. (2026). Rhino-orbito-cerebral fungal infection associated with mixed Cladosporium halotolerans and Penicillium corylophilum, complicated by internal carotid artery occlusion and ischemic stroke: a case report.. Journal of infection and chemotherapy : official journal of the Japan Society of Chemotherapy. https://doi.org/10.1016/j.jiac.2026.103007