• Christopher Polk, Carol Weida, Nikhil Patel, and Michael Leonard.
• Division of Infectious Diseases, Atrium Health, Charlotte, NC.
• Medicine (Baltimore). 2022 Sep 16; 101 (37): e30585.

IntroductionErdheim-Chester disease (ECD) commonly has neurologic manifestations but rarely presents with meningitis and hypoglycorrhachia. Here, were present a case of ECD with a clinical and laboratory presentation initially thought to be bacterial meningitis with sepsis.MethodsWe report a case of a 79-year-old with history of enigmatic bone pain and peritoneal nodules who presented with meningitis. After failure to improve on antibiotic therapy other etiologies of hypoglycorrhachia including sarcoid, tuberculosis, and fungal and carcinomatous meningitis were considered. However, no definite diagnosis could be made based on radiologic, serologic, microbiologic, and molecular testing and the patient failed to improve on empiric therapy including antibiotics, antifungals, and tuberculosis and steroid therapy.ResultsUltimately, autopsy revealed a new diagnosis of ECD manifesting as meningitis, a rare presentation of a rare disease.ConclusionAlthough only reported in one other case to our knowledge, ECD can present with meningitis with hypoglycorrhachia.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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