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Table of Contents    
CASE REPORT
Year : 2019  |  Volume : 67  |  Issue : 4  |  Page : 1107-1109

Solitary Intracerebral Phaeoid Fungal Granuloma: A Case Report


1 Department of Neurosurgery, IMS and Sum Hospital, Siksha ‘O’ Anusandhan University, Bhubaneswar, Odisha, India
2 Department of Pathology, IMS and Sum Hospital, Siksha ‘O’ Anusandhan University, Bhubaneswar, Odisha, India

Date of Web Publication10-Sep-2019

Correspondence Address:
Dr. Sureswar Mohanty
Department of Neurosurgery, IMS and Sum Hospital, Siksha ‘O’ Anusandhan University, Bhubaneswar - 751 003
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.266263

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 » Abstract 


Fungal granuloma in the brain parenchyma caused by pheohyphomycosis is extremely rare. Antifungal drugs are not very effective. The present report is a case of solitary pheohyphomycosis granuloma, which underwent surgical excision followed by antifungal drug treatment with excellent result.


Keywords: Antifungal drug, brain edema, fungal granuloma, pheohyphomycosis, raised ICP
Key Message: Although fungal granuloma of the brain are not uncommon pigmented fungal granuloma is quite rare.


How to cite this article:
Mohanty S, Mohanty P, Priyadarshini L. Solitary Intracerebral Phaeoid Fungal Granuloma: A Case Report. Neurol India 2019;67:1107-9

How to cite this URL:
Mohanty S, Mohanty P, Priyadarshini L. Solitary Intracerebral Phaeoid Fungal Granuloma: A Case Report. Neurol India [serial online] 2019 [cited 2019 Sep 23];67:1107-9. Available from: http://www.neurologyindia.com/text.asp?2019/67/4/1107/266263




Solitary pigmented fungal granuloma (phaeohyphomycosis) within brain parenchyma is extremely rare. Central nervous system (CNS) deep mycoses often occur as rhinocerebral zygomycosis, aspergillosis, abscess or acute or chronic meningitis by Candida, Cryptococcus, mucormycosis, coccidioides etc., and melanized fungi. In a review of 40 fungal granulomas collected from several centers in India,[1],[2] the most common was aspergillomas (25) followed by mucormycosis (12), cryptococcosis (3), Cladosporium (3), Bipolaris hawalarisis (1), and Candida species (1). However, no mention of pigmented hyphae or yeast-like cells in the granulomas was reported.[2]


 » Case Report Top


History of present illness

A 40-year-old female patient was apparently alright 5 days back. She developed a headache, vomiting (2–3 episodes), and inability to walk and talk. There was a history of blurring of vision since last 1 year; she was treated in the emergency department on 18-02-17 for altered sensorium and all the above symptoms. No history of diabetes mellitus, tuberculosis, and intake of the immunosuppressive drug was recorded. She had a history of a chronic discharging sinus on her hard palate which was treated at another hospital 2–3 years ago. No details of treatment were available. No significant family history was recorded. A history of taking a bath in a pond was reported. General examination was within normal limits.

Neurology examination revealed that the patient was conscious, not cooperative, and drowsy. She had mild right hemiparesis and ataxia and fundus retinal hemorrhages. The patient underwent magnetic resonance imaging (MRI) of the brain [Figure 1].
Figure 1: MRI revealed well-encapsulated thick-walled ring enhancing lesion with intense perilesional edema (could be granulomatous lesion, tuberculoma), and mass effect by within left frontal subcortical white matter

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Treatment

In view of the fast deteriorating consciousness, left frontal craniotomy based on the temporal muscle was done under general anesthesia. The dura was tense. It was opened with base towards the superior sagittal sinus through the mid frontal gyrus, and a 2-inch long cortiscectomy was done with cavitron ultrasonic aspirator (CUSA). A firm tumor was felt at a depth of 2-inch and the whole tumor was enucleated. Hemostasis was achieved. The brain was lax, and the dura was closed. The bone flap replaced and fixed with 2 pericranial stitches. The scalp was closed in two layers. The tumor was firm and sectioned by a sharp knife. The cut surface revealed colored tissue [Figure 2]. Postoperatively, the patient was administered intravenous voriconazole 200 mg for 1 week followed by oral voriconazole tablets for 6 weeks. Follow-up after 3 months revealed that the patient was intact and leading a normal life.
Figure 2: Surgical specimen on the cut section revealed soft-to-firm lesion colored with dark and brown pigments

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Pathology (gross and microscopy)

Grayish white globular soft-to-firm tissue with central cystic brownish area measuring 2 × 1.5 × 1.5 cm was noted. Microscopy revealed necrotizing granulomas with good numbers of foreign body type of giant cells engulfing the fungal elements and was reported as dematiaceous cerebral mycoses possibly due to pheohyphomycosis [Figure 3].
Figure 3: (a) Photomicrograph shows brown-colored (melanized) fungal hyphae with giant cell engulfing the fungal elements (H and E stain, 400×); (b) Photomicrograph showing necrotizing granulomas containing septate and branched hyphae with swollen wall (PAS stain, 400×)

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 » Discussion Top


According to the patient's history, she was habituated to pond bathing which might be the mode of transmission (through skin) of the causative organism. She also had a history of chronic discharging sinus on the hard palate for which she was treated 2–3 years back; which was also suspected as another route of transmission (through paranasal sinuses). Hematogenous spread is extremely rare,[3] yet a possible route of transmission in this case.

Pheohyphomycosis is a heterogeneous group of natural melanized opportunistic mycotic infections caused by dematiaceous fungi,[4] whose morphologic characteristics in the tissue include brownish short branched septate (2–6 μ width) hyphae and budding yeast-like cells.[5],[6] Many species are included such as Bipolaris, Cladophialophora bantiana,[7]Cladosporium, Exophiala, Fonsecaea pedrosoi, Phialophora, Ochronosis, Rhinocladiells, and Wangiella. These are typically soil organisms, decaying vegetable matter, and common laboratory contaminants; it is more commonly seen among immunocompromised patients through skin infections. Most common extremely neurotropic species is Xylohypha (Cladosporium) bantiana, which are confined to the brain and meninges. Cerebral lesions appeared encapsulated abscess [8] or generalized inflammatory infiltrates with granuloma formation.[6] An extensive survey of cerebral fungal granulomas has been reported from India.[9] However, phicoid phaeoid fungal granuloma is rarely reported.

The tissue is the mainstay for culture to identify the causative species. Dematiaceous fungi can frequently be discerned in tissue specimens stained with conventional hematoxylin and eosin; they appear as septate, brownish hyphae, or yeast-like cells, reflecting their high melanin content. Fungal stains such as Masson–Fontana, PAS, and Silver stains can also be used in tissue sections for identifying the specific morphology of fungus.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal the identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 » References Top

1.
Dubey A, Patwardhan RV, Sampth S, Vani S, Kolluri S, Nanda A. Intracranial fungal granuloma: Analysis of 40 patients and review of the literature. Surg Neurol 2005;63:254-60.  Back to cited text no. 1
    
2.
Ravisankar S, Chander RV. Cerebral pheohyphomycosis: Report of a rare case with review of literature. Neurol India 2013;61:526-8.  Back to cited text no. 2
  [Full text]  
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.
Horre R, de Hoog GS. Primary cerebral infections by melanized fungi: A review. Stud Mycol 1999;43:176-93.  Back to cited text no. 4
    
5.
Zarrin M, Mahmoudabadi AZ. Central nervous system fungal infections; a review article. Jundishapur. J Microbiol 2010;3:41-7.  Back to cited text no. 5
    
6.
Shankar SK, Mahadevan A, Sundaram C, Sarkar C, Chacko G, Lanjewar DN, et al. Pathobiology of fungal infections of the central nervous system with special reference to the Indian scenario. Neurol India 2007;55:198-215.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Suri P, Chhina DK, Kaushal V, Kaushal RK, Singh J. Cerebral Phaeohyphomycosis due to Cladophialophora bantiana – A Case Report and Review of Literature from India. J Clin Diagnostic Res 2014;8:DD01-5.  Back to cited text no. 7
    
8.
Jung Na-Y, Kim E. Cerebral Phaeohyphomycosis: A Rare Cause of Brain Abscess. J Korean Neurosurg Soc 2014;56:444-7.  Back to cited text no. 8
    
9.
Sharma BS, Khosla VK, Kak VK, Banerjee AK, Vasishtha RK, Prasad KS, et al. Intracranial fungal granuloma. Surg Neurol 1997;47: 489-97.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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