Curvularia Abscess of the Brainstem Branko Skovrlj 1 , Maryam Haghighi 2 , Mark E. Smethurst 3 , John Caridi 1 , Joshua B. Bederson 1 INTRODUCTION Curvularia is a filamentous, dematiaceous fungus characterized by melanin pigmen- tation in the cell walls of its hyphae (22). It is a mold, ubiquitously found in soil around the world, with preference to the tropical and subtropical regions (20). Infections by dematiaceous fungi, also known as phaeo- hyphomycosis, are rare in humans (25). Curvularia was first documented as a human pathogen in 1959 in Africa, when it was isolated from a lung mycetoma (2, 4, 9). The first central nervous system (CNS) Curvularia infection was described in 1977 by Lampert et al. (19). Since then, only eight CNS Cur- vularia infections have been documented in the literature (Table 1) (4, 8, 11, 14, 15, 19, 23, 26, 29, 30). CASE REPORT A 33-year-old African-American man with past medical history of occasional migraine headache presented to the emer- gency room with worsening right-sided headache and intermittent nausea with unpredictable episodes of severe vomiting. He complained of left-sided paresthesias, gait imbalance, blurry vision, diplopia, and erectile dysfunction for a period of 2 weeks. He described a recent 20-lb weight loss and progressive exercise intolerance. His social history was significant for 20 years of heavy marijuana use as well as significant alcohol use. Physical examination findings included a temperature of 96.8  F, blood pressure of 129/65 mm Hg, and heart rate of 103 beats per minute. Neurological examination dis- closed right-sided ptosis, left hemisensory loss, leftward deviation of the uvula, right tongue deviation, nystagmus on rightward gaze, and a positive Romberg sign. Labora- tory findings included a normal complete blood count and electrolyte panel. Cerebro- spinal fluid examination revealed increased cellularity of polymorphonuclear leukocytes and lymphocytes. The cerebrospinal fluid culture was negative. Magnetic resonance imaging (MRI) of the brain demonstrated a 1.6  1.6  1.7 cm right-sided, homoge- neously enhancing lesion within the right medulla with surrounding mass effect causing expansion of the brainstem at this location and vasogenic edema extending into the pons and upper cervical spinal cord (Figure 1). A computed tomography scan of the chest revealed a left lung lesion, which on subsequent biopsy was positive for multinucleated giant cells consistent with a diagnosis of granuloma, yielding no further clues to the etiology of the brainstem lesion. Due to the lack of diagnosis as well as the patient’s deteriorating neurological status, a decision was made to perform an open biopsy and lesion resection. A preop- erative MRI of the brain performed several days later revealed significant changes, - OBJECTIVE: To present a unique case of a brainstem Curvularia fungal infection and review the diagnosis and management of this rare phenomenon. - METHODS: A 33-year-old immunocompetent African American male presented with 2 weeks of headache, nausea, and vomiting in a setting of a recent 20-lb weight loss. Neurological examination was positive for multiple cranial nerve palsies, hemisensory loss, and gait instability. Magnetic resonance imaging demonstrated an enhancing medullary lesion. - RESULTS: Metastatic and infectious workup revealed a left lung lesion, which on subsequent biopsy was positive for a granuloma yielding no further clues to the etiology of the brainstem lesion. On surgical exploration of the cranial lesion, a puss-filed, encapsulated lesion was encountered that was tightly adherent to the brainstem. Intraoperative biopsy of the lesion capsule was initially negative but on postoperative day 9, fungal hyphae were encountered identified on morphology as Curvularia species. The patient was started on triple antifungal therapy but necessitated a second surgery for lesion debulking and drainage. The patient was discharged home 10 weeks after initial presentation. At the 13-months follow-up the patient is doing very well and his neurological examination continues to improve. - CONCLUSIONS: This is the first reported case of a brainstem Curvularia infection. This case highlights the importance of an aggressive surgical and antibiotic therapy in the treatment of central nervous system Curvularia infec- tions. There appears to be a strong relationship between heavy marijuana use and Curvularia infection, producing lung granulomas that may extend to other organs such as the central nervous system of immunocompetent patients. Key words - Brainstem - Curvularia abscess - Fungal central nervous system infection - Phaeohyphomycosis Abbreviations and Acronyms CNS: Central nervous system MRI: Magnetic resonance imaging From the 1 Department of Neurosurgery, Mount Sinai School of Medicine, New York City, New York; 2 School of Medicine, Karolinska Institutet, Hagalund, Sweden; and 3 Department of Pathology, James J. Peters VA Medical Center, Bronx, New York, USA To whom correspondence should be addressed: Branko Skovrlj, M.D. [E-mail: skovrlj.branko@gmail.com] Citation: World Neurosurg. (2014) 82, 1/2:241.e9-241.e13. http://dx.doi.org/10.1016/j.wneu.2013.07.014 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2014 Elsevier Inc. All rights reserved. WORLD NEUROSURGERY 82 [1/2]: 241.e9-241.e13, JULY/AUGUST 2014 www.WORLDNEUROSURGERY.org 241.E9 Peer-Review Short Reports