Recurrent bipolaris hawaiiensis infection presenting as an intracranial mass lesion

Saba S 1, Montero J 2, Rosenbach K 3, Kulkarni V 4

Full title: 

Recurrent bipolaris hawaiiensis infection presenting as an intracranial mass lesion

Abstract: 

Bipolaris, a dematiaceous filamentous fungus often isolated from plant materials, is known to cause granulomatous disease in cattle. Bipolaris is one of the causative agents of human phaeohyphomycosis. Three species, B. spicifera, B. australiensis, and B. hawaiiensis are associated with human disease. The clinical spectrum is diverse, including allergic and chronic invasive sinusitis, meningoencephalitis, pulmonary infections, and allergic bronchopulmonary disease. Bipolaris can infect both immunocompetent and immunocompromised hosts. Invasive sinusitis and allergic fungal sinusitis is thought to occur after inhalation of the organism. B. spicifera has been reported to cause fungus balls and locally invasive rhinosinusitis. B. hawaiiensis has also been reported to cause fungal sinusitis, along with intracranial extension via erosion of the cranial base. Interestingly, invasive disease has been seen primarily in patients with intact immune systems. Treatment has generally consisted of local resection followed by intravenous amphotericin B followed by a prolonged course of itraconazole. We report a case of an immunocomptetent African American male presenting with a fungating mass in his brain initially thought to be neoplastic process. The clinical presentation included a gradual alteration of his mental status over a period of weeks to months with loss of motor, speech, and inhibitory control including inappropriate behavior. MRI of the brain revealed a large fungating, penetrating lesion in the frontal lobe of his brain. Histology was consistent with fungal etiology, and initially thought to be Blastomycoses. However, cultures yielded a black mold, and were not consistent with the histologic diagnosis. Initially, the speciation of the fungus was in doubt due to the organism’s time requirement for sporulation. Previous medical records from an outside institution, indicated the patient had a similar mass resected from his brain two years earlier. At that time, cultures identified the black mold as Bipolaris hawaiiensis. By this time our microbiology lab had also confirmed the species as B. hawaiiensis. During his previous hospitalization he was treated with itraconazole alone. It was unclear from previous records the duration of his treatment, but he apparently had stopped taking itraconazole at least 6 months prior to this hospitalization. We treated the patient with intravenous amphotericin B lipid complex and discharged him with oral voriconazole for one year. This case not only demonstrates the presentation of an unusual infection of the central nervous system with a filamentous dematiaceous fungus, but highlights the difficulty in obtaining a rapid accurate diagnosis of the species involved. In addition, it is noteworthy to realize that recurrence can occur despite initial adequate therapy if the duration of therapy is not long enough to eradicate the organism. References: 1. Fryen A, Mayser P, Glanz F, et al. Allergic fungal sinusitis caused by Bipolaris (Drechslera) hawaiiensis. Eur Arch Otolorhinolaryngol 1999;256(7):330-4 2. Filizzola MJ, Martinez F, Rauf SJ. Phaeohyphomycosis of the central nervous system in immunocompetent hosts: report of case and review of literature. Int J Infect Dis. 2003;7(4):282-6 3. Revankar SG, Patterson JE, Sutton DA, et al. Disseminated phaeohyphomycosis: review of an emerging mycosis. Clin Inf Dis. 2002;34:467-76 4. Flanagan KL, Bryceson ADM. Disseminated infection due to Bipolaris australiensis in a young immunocompetent man: case report and review. Clin Inf Dis. 1997; 25:311-3 5. Iwen PC, rupp ME, Hinrichs SH. Invasive mold sinusitis: 17 cases in immunocompromised patients and review of the literature. Clin inf Dis. 1997;24:1178-84
2005

abstract No: 

64

Full conference title: 

15th Annual Focus on Fungal Infections