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CASE REPORT
Year : 2015  |  Volume : 4  |  Issue : 1  |  Page : 42-44

A rare case of cerebral pheohyphomycosis in an immunocompetent patient from central India


1 Department of Microbiology, Sri Aurobindo Institute of Medical Sciences Medical College and Post Graduate Institute, Indore, India
2 Department of Pathology, Sri Aurobindo Institute of Medical Sciences Medical College and Post Graduate Institute, Indore, India
3 Department of Pathology, Gagrani Hospital, Dewas, Madhya Pradesh, India

Date of Web Publication13-Jan-2015

Correspondence Address:
Trupti Bajpai
Department of Microbiology, Sri Aurobindo Institute of Medical Sciences Medical College and Post Graduate Institute, Indore - 453 555, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-344X.149264

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  Abstract 

A 45-year-old immunocompetent man, tailor by occupation, presented with pyrexia of unknown origin since last 6 months. He was diagnosed to be a case of cerebral pheohyphomycosis caused by dematiceous fungi, Cladophylophora bantiana. Unfortunately, the patient succumbed to death before the confirmation of diagnosis. To conclude, apart from tubercular meningitis, cerebral pheohyphomycosis should be considered in differential diagnosis while examining the immunocompetent patients exhibiting chronic features of pyrexia of unknown origin.

Keywords: Cerebral pheohyphomycosis, Cladophylophora, dematiceous fungi


How to cite this article:
Bajpai T, Nandedkar S, Bhatambare GS, Gagrani N. A rare case of cerebral pheohyphomycosis in an immunocompetent patient from central India. Int J Health Allied Sci 2015;4:42-4

How to cite this URL:
Bajpai T, Nandedkar S, Bhatambare GS, Gagrani N. A rare case of cerebral pheohyphomycosis in an immunocompetent patient from central India. Int J Health Allied Sci [serial online] 2015 [cited 2022 Jan 25];4:42-4. Available from: https://www.ijhas.in/text.asp?2015/4/1/42/149264


  Introduction Top


Cerebral pheohyphomycosis is a rare and frequently fatal clinical syndrome diagnosed in patients with deep-seated cerebral infections. It is a heterogenous group of mycotic infection caused by dematiceous fungi. Such fungi are frequently known for their neurotropism, although central nervous system (CNS) seeding may occur through inoculation in the skin of extremities following even a minor trauma. [1] CNS pheohyphomycosis presents with unusual features and is associated with poor prognosis irrespective of the immune status of patient. [2],[3]

Ours is a case of cerebral pheohyphomycosis from central India, presented by an immunocompetent man with suspected history of trauma. Unfortunately, it could not be managed successfully and by the time we reached the confirmed diagnosis, the patient succumbed to death.


  Case report Top


A 45-year-old man, tailor by occupation, was admitted to our hospital with complain of chills, high grade fever, and irritability since last 3 days while heaviness in chest, bodyache, and dyspnea since last 2 days. He presented with the history of on and off fever since last 6 months and burning sensation all over the body since last 4 months.

On examination, he was conscious, oriented, and presented no signs of meningitis. He did not present any history of diabetes mellitus, hypertension, chest problems, and pulmonary tuberculosis. His hematological and biochemical parameters were within normal limits. Considering the pyrexia of unknown origin (PUO), his blood was serologically tested for the presence of malaria parasite (MP) and Widal, both of which tested negative. His blood culture was negative. He was also negative for human immunodeficiency virus (HIV) and hepatitis B surface antigen (HBsAg). Lumbar tap resulted into a xanthochromic and clear cerebrospinal fluid (CSF) [Figure 1]a with a protein concentration of 160 mg/dl, glucose concentration of 30 mg/dl, and 82 leukocytes/mm 3 among which 60% were lymphocytes and 40% were neutrophils. CSF adenosine deaminase (ADA) value was 72 IU/L and was negative for acid fast bacilli (AFB).
Figure 1: (a) Xanthochromic cerebrospinal fluid (CSF) (b) Giemsa-stained cytology slide of cerebrospinal fluid (CSF) revealing fungal hyphae [1000x]

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His admission work-up also included computed tomography (CT) scan that revealed only the dilation of ventricles that probably indicated the early signs of meningitis. No lesions in the cerebrum were evident. Based on the above findings, it was thought to be a case of tubercular meningitis.

Giemsa stained cytology slide of CSF revealed occasional fungal hyphae [Figure 1]b. On subjecting it to culture on 4% Sabaurauds Dextrose Agar (SDA) (Hi Media, Mumbai, India) with chloramphenicol (50 mg/L) and incubation at 42 o C revealed velvety, olive gray to brown colonies on obverse and dark tan on the reverse of SDA slant in nine days of incubation [Figure 2] a and b Potassium hydroxide (KOH) preparation of the growing fungus revealed brown-pigmented hyphal elements [Figure 2]c Lactophenol cotton blue preparation revealed conidiophores that were indistinguishable from the vegetative hyphae. One-celled blastoconidia borne in long, sparsely branching chains were prominent. [Figure 2]d.
Figure 2: (a) Obverse of Sabaurauds Dextrose Agar (SDA) slant showing olive gray to brown colonies (b) Reverse of Sabaurauds Dextrose Agar (SDA) slant showing dark tan (c) Brown pigmented fungus in potassium hydroxide (KOH) preparation [400x] (d) Lactophenol cottonblue (LCB) preparation showing conidiophores and branching conidia [400x]

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The etiological agent was identified as Cladophilophora bantiana (Xylophila bantiana) and the final impression of cerebral pheohyphomycosis was given. Unfortunately, the patient died on the next day of CSF tap.


  Discussion Top


Cerebral pheohyphomycosis is a general term applicable to all those brain infections where etiological agent is a dematiaceous fungus. [4] Out of the various species implicated in cerebral pheohyphomycosis, most of them cause secondary infections following development of infection in other sites (mostly sinuses). Only four species like Cladophialophora bantiana, Exophiala dermatitidis, Ochroconis gallopavum, and Ramichloridium mackenziei are known to have a high neurotropic potential. [1],[5] C. bantiana accounts for the majority of cases documented till date; [6],[7] and in our case also, the etiological agent was C. bantiana. The fungus is known by several names such as Cladosporium trichoides, Xylohypha bantiana, and Cladosporium bantianum.[1],[8]

Though the portal of entry is not well-established, inhalation of spores followed by colonization and subsequent hematogenous spread has been widely suggested. [2] Contiguous spread through paranasal sinuses or trauma resulting into implantation of fungal elements into skin and its subsequent dissemination may be the cause of disease. [6] Though, in our case, patient did not present any history of trauma, but we strongly suspect minor trauma that resulted into implantation of fungal elements into his skin. We would correlate it with patient's tailoring occupation just like several other studies that have also correlated the cause of infection with the patient's occupation. [1],[4],[6] Though, the life-threatening fungal infections are often associated with severe immuno-compromised status, a typical but confounding and peculiar feature of this infection is that, it is increasingly recognized in immunocompetent persons with no underlying diseases. [1],[2],[9] The disease has also been frequently reported in males and in our case also the patient was an immunocompetent male. [1],[2],[4],[6],[9]

Radiological investigations are always helpful in reaching the diagnosis. In our case, computed tomography (CT) scan was suggestive of early signs of meningitis but could not provide any special clues. Patients with cerebral pheohyphomycosis usually present with brain abscess and histopathological findings play an important role in diagnosis of disease. Our patient presented with early signs of meningitis only, which has been observed by few authors in their studies. [1] Xanthochromia in CSF is likely to be because of basal meningitis, as mild lateral ventricular dilation was seen in CT. Out of the several reasons responsible for xanthochromia, our case involved a high level of CSF protein (160 mg%) and literature reveals that xanthochromia results if there is more than 150 mg% or more protein, depending upon the proportion of chromogens. [10] Our study reported the presence of occasional fungal elements in spine fluid. Similar findings have been made by few authors. [11] Cytological examination of the causative organism is not enough to determine the type of fungus. Hence, second step must be to demonstrate the cultural aspects of the causative fungus. [3] In our case, we confirmed the etiological agent both through cytological as well as microbiological investigations.

Treatment has largely been unsuccessful in most of the cases of cerebral pheohyphomycosis. It must be individualized with surgical resection and optimized anti-fungal therapy. [2],[5],[9] We could not plan any treatment for our patient as he succumbed by the time we reached our final diagnosis.

To conclude, apart from tubercular meningitis, cerebral pheohyphomycosis should be considered in differential diagnosis while examining the immunocompetent patients exhibiting chronic features of pyrexia of unknown origin. Also their CSF specimen should be cytologically and microscopically examined for the presence of fungal elements. Such specimen should be handled with biosafety level 2 containment because of the known pathogenesis of these organisms for immunocompetent individuals. Quick decisions, radiodiagnosis and culture reports can increase the feasibility of surgical resection in the patients of cerebral pheohyphomycosis and antifungal treatment can be very well-individualized using radiological evidences of resolution.

 
  References Top

1.
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. 1
    
2.
Ravishankar 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
    
3.
Suri P, Chhina DK, Kaushal V, Kaushak RK, Singh J. Cerebral Pheohyphomycosis due to Cladophialophora bantiana- A case report and review of literature from India. J Clin Diagn Res 2014;8:DD01-5.  Back to cited text no. 3
    
4.
Nóbrega JP, Rosemberg S, Adami AM, Heins-Vaccari EM, Lacaz Cda S, de Brito T. Fonsecaea pedrosoi cerebral phaeohyphomycosis ("chromoblastomycosis"): First human culture-proven case reported in Brazil Rev Inst Med Trop Sao Paulo 2003;45:217-20.  Back to cited text no. 4
    
5.
Tello J, Otal T. Cerebral phaeohyphomycosis caused by fonsecaea monophora in a renal transplant patient. Infect Med J 2008; 25:469-73.  Back to cited text no. 5
    
6.
Chugh A, Rane S, Gandhoke C, Maithili K. A rare case report of cerebral phaeohyphomycosis mimicking glioma in a patient of rheumatic valvular heart disease. Indian J Neurosurg 2013;3:199-202.  Back to cited text no. 6
    
7.
Heney C, Song E, Kellen A, Raal F, Miller SD, Davis V. Cerebral pheohyphomycosis caused by Xylohyphabantiana. Eur J Clin Microbiol Infect Dis 1989;8:984-8.  Back to cited text no. 7
    
8.
Chander J. Textbook of medical mycology. Pheohyphomycosis. 2 nd ed. Ch. 14. p. 147, Mehta publishers, New Delhi; 2002.  Back to cited text no. 8
    
9.
Palaoglu S. A rare fungal brain abscess caused by Cladosporium trichoides. A review of the literature. Turk Neurosurg 1989;1:26-9.  Back to cited text no. 9
    
10.
Cohen J, Fadul C, Jenkyn L, Ward T. Disorders of the Nervous system. In: Swenson R, Reeves A, editors. Neurologic Tests. Ch. 11. Dartmouth, 2008.  Back to cited text no. 10
    
11.
Bell WR, Dalton JB, McCall CM, Karram S, Pearce DT, Memon W et al. Iatrogenic Exserohilum infection of the central nervous system: Mycological identification and histopathological findings. Mod Pathol 2013;26:166-70.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2]


This article has been cited by
1 An updated comprehensive systematic review of Cladophialophora bantiana and analysis of epidemiology, clinical characteristics, and outcome of cerebral cases
A. Serda Kantarcioglu,Josep Guarro,Sybren De Hoog,Hulya Apaydin,Nuri Kiraz
Medical Mycology. 2016; : myw124
[Pubmed] | [DOI]



 

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