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LETTER TO EDITOR
Year : 2014  |  Volume : 62  |  Issue : 5  |  Page : 551-552

A case of cerebral pheohyphomycosis in an immunocompetent patient: Emphasis on intraoperative findings


1 Department of Neurosurgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
2 Department of Histopathology, Post Graduate Institute of Medical Education and Research, Chandigarh, India

Date of Submission20-Aug-2014
Date of Decision20-Aug-2014
Date of Acceptance25-Sep-2014
Date of Web Publication12-Nov-2014

Correspondence Address:
Ashish Aggarwal
Department of Neurosurgery, Post Graduate Institute of Medical Education and Research, Chandigarh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.144461

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How to cite this article:
Aggarwal A, Salunke P, Mohindra S, Garg R, Radotra B D. A case of cerebral pheohyphomycosis in an immunocompetent patient: Emphasis on intraoperative findings. Neurol India 2014;62:551-2

How to cite this URL:
Aggarwal A, Salunke P, Mohindra S, Garg R, Radotra B D. A case of cerebral pheohyphomycosis in an immunocompetent patient: Emphasis on intraoperative findings. Neurol India [serial online] 2014 [cited 2019 Sep 19];62:551-2. Available from: http://www.neurologyindia.com/text.asp?2014/62/5/551/144461


Sir,

Cerebral pheohyphomycosis is a fungal infection caused by dematiaceous septate mycelial elements and is associated with a very high mortality unless diagnosed and treated early. [1] It is extremely difficult to diagnosis this fungal infection even with newer radiological modalities.

A 42-year-old male, cook by profession and apparently immunocompetent presented with headache and altered sensorium of 2 weeks duration. Examination was unremarkable except for bilateral mild papilledema. Magnetic resonance imaging (MRI) showed a T1-weighted hypointense lesion in right parietal region. The lesion was intensively contrast enhancing with multiple small lesions surrounding the main lesion [Figure 1]. It was hyperintense on T2-weighted sequence and there was restriction on diffusion-weighted imaging (DWI) [Figure 1]. In view of altered sensorium he was taken up for surgery with a preoperative diagnosis of high grade malignancy. During surgery, aspiration of the lesion showed pus. The abscess wall was removed in total after defining a clear plane of cleavage between brain and abscess. The wall of abscess had multiple blackish spots. The frozen section of abscess wall revealed granulomas and multinucleated giant cells containing pigmented fungi. Loading dose of amphotericin B was given intraoperatively. Biopsy report confirmed the diagnosis of fungal infection, pheohyphomycosis [Figure 2]. Patient has received 1,500 mg. of amphotericin B and is on oral voriconazole, doing well at follow-up.
Figure 1: (a) NCCT head showing hypodense lesion in right parietooccipital region with surrounding perilesional edema. (b) MRI plain T1WI axial showing hypointense lesion and (c) ring enhancement on contrast. (d) MRI T2WI axial showing hyperintense lesion with perilesional edema. (e) MRI contrast T1WI sagittal showing ring enhancing lesion and a few satellite lesions. (f) MRI FLAIR coronal image showing almost isointense lesion with surrounding edema. (g) MRI DWI showing restriction of diffusion and (h) postoperative NCCT head showing complete excision of lesion. NCCT = Noncontrast computerized tomography, MRI = magnetic resonance imaging, T1WI = T1-weighted imaging, T2WI = T2-weighted imaging, FLAIR = fluid-attenuated inversion recovery, DWI = diffusion-weighted imaging

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Figure 2: (a) Microphotograph shows septate hyphae (curved arrow) engulfed by giant cells (straight arrow). The fungal elements are black in color. (a) Hematoxylin and eosin stain (×40) and (b) PAS stain ( ×40). PAS = Periodic acid-Schiff

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The term pheohyphomycosis means dark pigmented fungal hyphae. [2] This infection is caused in majority of cases by a single order Chaetothyriales. About 50% patients of cerebral pheohyphomycosis are immunocompetent. [3] Various theories regarding the spread to brain have been proposed: Hematogenous dissemination, spread through lymphatic vessels, contiguous spread from adjacent organs such as orbits/sinuses. [1] Clinical presentation is like that of any other space occupying lesion with a shorter duration of symptoms than tumor. Classically described MRI findings are ring enhancing lesions on T1-weighted imaging, hypointensity of the rings on T2-weighted imaging, and low to high signal on DWI. [1] We were misled to the preoperative diagnosis of high-grade glioma in the present case because of absence of fever and other constitutional symptoms of infection and hyperintensity on T2-weighted imaging. Intraoperative frozen section is of paramount importance in such cases. This helps in instituting loading dose of antifungal intraoperatively. Any delay in starting antifungal therapy may affect the outcome.

The mortality of untreated cases of pheohyphomycosis is near 100% and with surgery and antifungal agents it is about 65%. [4],[5] Because of this high mortality, a high index of suspicion is essential. Once suspected an urgent stereotactic guided aspiration of pus and fungal staining and culture should be the treatment option. In case the intraoperative findings raise the suspicion of fungal infection especially in the absence of typical clinicoradiological features, first dose of antifungal may be considered. A frozen section/crush smear done at the time of surgery immensely aids in diagnosis and management of cerebral fungal infections.

 
  References Top

1.
Li DM, de Hoog GS. Cerebral phaeohyphomycosis-a cure at what lengths? Lancet Infect Dis 2009;9:376-83.  Back to cited text no. 1
    
2.
Ajello L, Georg LK, Steigbigel RT, Wang CJ. A case of phaeohyphomycosis caused by a new species of Phialophora. Mycologia 1974;66:490-8.  Back to cited text no. 2
    
3.
Revankar SG, Sutton DA, Rinaldi MG. Primary central nervous system phaeohyphomycosis: A review of 101 cases. Clin Infect Dis 2004;38:206-16.  Back to cited text no. 3
    
4.
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;43:91-5.  Back to cited text no. 4
    
5.
Amar SS, Al-Tawfiq JA. Aspiration cytology of brain abscess from a fatal case of cerebral phaeohyphomycosis due to Ramichloridium mackenziei. Diagn Cytopathol 2007;35:695-9.  Back to cited text no. 5
    


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