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Acute Stroke due to Cunninghamella bertholletiae orbital cellulitis - a case report and review of the literature

Christoph Oster, Julia Kristina Steinborn, Tobias Blau, Peter Michael Rath, Kurt Werner Schmid, Cornelius Deuschl, Christoph Kleinschnitz, Andreas Totzec k

Introduction: We present a case with repeated cerebral infarction by thrombotic fungal mycelium (Cunninghamella bertholletiae) in the right and left pre-stenosed internal carotid artery.

Case: A 72-year-old female patient with a myelodysplastic syndrome presented with a right orbital swelling and a loss of vision. A computer tomography scan and magnetic resonance imaging were suspicious of orbital cellulitis. An orbital decompression was performed. After surgery, the patient developed a hemiparesis on the left side. A computer tomography revealed an internal carotid artery occlusion on the right side. A thrombectomy was performed successfully. The following days a central artery occlusion on the right eye was diagnosed. A lumbar puncture revealed a highly increased white blood count matching to the diagnosis of a meningoencephalitis. On the fourth day of treatment in the neurology department, the patient developed fixed and dilated pupils. A computer tomography showed new bilateral anterior cerebral artery infarctions and infarction of the entire territory of the left middle cerebral artery. Intensive care treatment was terminated. The autopsy found an orbital cellulitis with invasive mycosis caused by mould. Internal transcribed spacer (ITS) sequencing revealed Cunninghamella bertholletiae in the material obtained from the orbit and the left internal carotid artery. Retrospectively, sepsis of thrombotic fungal mycelium due to increased pathological coagulation repeatedly resulted in arterial embolism.

Conclusion: In immunosuppressed patients, rare pathogens can cause sepsis and septic complications. A calculated antifungal therapy should be considered in cryptic cases of meningoencephalitis.