Cerebral phaeohyphomycosis due to Rhinocladiella mackenziei in an immunocompetent patient: A case report and review of literature

Document Type : Case report

Authors

Department of Medicine, Aga Khan University Hospital, Stadium Road, Karachi, Pakistan

Abstract

Background and Purpose: Rhinocladiella mackenziei is a neurotropic fungus, which can cause devastating intracerebral infections with up to 100% fatality rate. It is difficult to isolate this fungus in laboratory as it grows slowly and requires diagnostic skills.
Case report: A 42-year-old Pakistani man presented with headache, facial numbness, progressive upper limb weakness, and dysarthria. Magnetic resonance imaging of the brain showed a space-occupying lesion in the basal ganglia region. The patient underwent supratentorial craniotomy for biopsy and excision. Histopathology of the specimen revealed granulomatous inflammation with abscess formation. Periodic acid-Schiff special stains highlighted the presence of numerous septate fungal hyphae. The results revealed the growth of dematiaceous fungi, which were morphologically classified as R. mackenziei. The patient is currently stable and is being on amphotericin and posaconazole, along with neurorehabilitation therapy.
Conclusion: Rhinocladiella mackenziei brain abscess is a devastating infection with significant mortality. This condition should be suspected in patients with brain abscess from high endemic areas.
 

Keywords


1. Yusupov N, Merve A, Warrell CE, Johnson E, Curtis C, Samandouras G. Multiple brain abscesses caused by Rhinocladiella mackenziei in an immunocompetent patient: a case report and literature review. Acta Neurochirur. 2017; 159(9):1757-63.
2. Li DM, de Hoog GS. Cerebral phaeohyphomycosis--a cure at what lengths? Lancet Infect Dis. 2009; 9(6):376-83.
3. Al-Tawfiq JA, Boukhamseen A. Cerebral phaeohyphomycosis due to Rhinocladiella mackenziei (formerly Ramichloridium mackenziei): case presentation and literature review. J Infect Public Health. 2011;
4(2):96-102.
4. Moreno LF, Ahmed AA, Brankovics B, Cuomo CA, Menken SB, Taj-Aldeen SJ, et al. Genomic understanding of an infectious brain disease from the desert. G3 (Bethesda). 2018; 8(3):909-22.
5. Mirhendi H, Fatemi MJ, Bateni H, Hajabdolbaghi M, Geramishoar M, Ahmadi B, et al. First case of disseminated phaeohyphomycosis in an immunocompetent individual due to Alternaria malorum. Med Mycol. 2013; 51(2):196-202.
6. Mohammadi R, Mohammadi A, Ashtari F, Khorvash F, Hakamifard A, Vaezi A, et al. Cerebral phaeohyphomycosis due to Rhinocladiella mackenziei in Persian Gulf region: a case and review. Mycoses. 2018; 61(4):261-5.
7. Cristini A, Garcia-Hermoso D, Celard M, Albrand G, Lortholary O. Cerebral phaeohyphomycosis caused by Rhinocladiella mackenziei in a woman native to Afghanistan. J Clin Microbiol. 2010; 48(9):3451-4.
8. Jabeen K, Farooqi J, Zafar A, Jamil B, Mahmood SF, Ali F, et al. Rhinocladiella mackenziei as an emerging cause of cerebral phaeohyphomycosis in Pakistan: a case series. Clin Infect Dis. 2011; 52(2):213-7.
9. Chowdhary A, Meis JF, Guarro J, de Hoog GS, Kathuria S, Arendrup MC, et al. ESCMID and ECMM joint clinical guidelines for the diagnosis and management of systemic phaeohyphomycosis: diseases caused by black fungi. Clin Microbiol Infect. 2014; 20(Suppl 3):47-75.
10. Revankar SG. Phaeohyphomycosis. Infect Dis Clin North Am. 2006; 20(3):609-20.
11. Hardman N, Young N, Hobson R, Sandoe J, Wellberry-Smith M, Thomson S, et al. Prolonged survival after disseminated Rhinocladiella infection treated with surgical excision and posaconazole. Transpl Infect Dis. 2020; 22(2):e13264.
12. Gaviani P, Schwartz RB, Hedley-Whyte ET, Ligon KL, Robicsek A, Schaefer P, et al. Diffusion-weighted imaging of fungal cerebral infection. AJNR Am J Neuroradiol. 2005; 26(5):1115-21.
13. Al-Abdely HM, Alkhunaizi AM, Al-Tawfiq JA, Hassounah M, Rinaldi MG, Sutton DA. Successful therapy of cerebral phaeohyphomycosis due to Ramichloridium mackenziei with the new triazole posaconazole. Med Mycol. 2005; 43(1):91-5.
14. Pitisuttithum P, Negroni R, Graybill JR, Bustamante B, Pappas P, Chapman S, et al. Activity of posaconazole in the treatment of central nervous system fungal infections. J Antimicrob Chemother. 2005; 56(4):745-55.
15. Deng S, Pan W, Liao W, de Hoog GS, Gerrits van den Ende AH, Vitale RG, et al. Combination of amphotericin b and flucytosine against neurotropic species of melanized fungi causing primary cerebral phaeohyphomycosis. Antimicrob Agents Chemother. 2016; 60(4):2346-51.
16. Taj-Aldeen SJ, Almaslamani M, Alkhalf A, Al Bozom I, Romanelli AM, Wickes BL, et al. Cerebral phaeohyphomycosis due to Rhinocladiella mackenziei (formerly Ramichloridium mackenziei): a taxonomic update and review of the literature. Med Mycol. 2010; 48(3):546-56.
17. Khaliq MF, Ihle RE, Schirtzinger CP. Cladophialophora bantiana cerebral phaeohyphomycosis complicated by pulmonary nocardiosis: a tale of two infections. Case Rep Infect Dis. 2019; 2019:4352040.
Volume 6, Issue 3
September 2020
Pages 65-68
  • Receive Date: 13 March 2020
  • Revise Date: 08 September 2020
  • Accept Date: 12 September 2020
  • Publish Date: 01 September 2020