A Rare Case of Central Nervous System Mucormycosis Following Deep Brain Stimulation Implantation

Introduction: Central nervous system (CNS) mucormycosis is a severe intracranial fungal infection with mortality exceeding 50%, usually occurring in patients with immunosuppression or severe comorbidities. We present a rare case of delayed-onset CNS mucormycosis occurring four years post-deep brain stimulation (DBS) implantation in an immunocompetent patient with Parkinson’s disease.


Description: A 46-year-old woman with a 7-year history of Parkinson’s disease underwent DBS implantation 4 years prior, achieving stable disease control without any history of immunosuppression. She presented with persistent fever for two weeks and leukocytosis, with no response to empirical cephalosporins. Her condition rapidly deteriorated, with body temperature peaking at 40.9°C, accompanied by slurred speech and progressive respiratory distress. Head CT revealed mild cerebral edema. Despite administration of corticosteroids and hyperosmolar agents, she suffered a cardiac arrest and was transferred to our ICU after successful resuscitation. Subsequent head CT showed worsening cerebral edema, and sinus CT demonstrated extensive bilateral maxillary, ethmoidal, and sphenoidal sinusitis. Metagenomic next-generation sequencing (mNGS) detected Rhizopus (922 reads) and Mucor (610 reads) in cerebrospinal fluid (CSF), as well as low-level Mucorales DNA in peripheral blood. Immediate antifungal therapy with liposomal amphotericin B and isavuconazole was initiated alongside antibiotics. Even with intensive antifungal and supportive treatment, the patient developed uncal herniation and progressed to irreversible deep coma with fixed, dilated pupils, absent light reflex, and electroencephalography confirmed severe brain injury. Given the poor prognosis, the family decided to transition care to comfort measures.


Discussion: This case highlights the importance of maintaining clinical suspicion for CNS mucormycosis in post-DBS implantation patients presenting with unexplained fever, even years after implantation and without immunosuppression. Early mNGS-based CSF testing enables timely diagnosis and facilitates the early initiation of amphotericin B–based antifungal therapy with close toxicity monitoring. Heightened awareness of this rare but lethal complication is essential for prompt intervention and improved prognosis.