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 ~  Abstract
 ~ Introduction
 ~ Case Report
 ~  Specific treatme...
 ~ Discussion
 ~ Acknowledgment
 ~  References
 ~  Article Figures

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  Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 32  |  Issue : 4  |  Page : 440-442
 

Cerebral phaeohyphomycosis - could early diagnosis have saved the patient?


Department of Microbiology, Government Medical College, Nagpur, Maharashtra, India

Date of Submission19-Sep-2013
Date of Acceptance21-Mar-2014
Date of Web Publication4-Oct-2014

Correspondence Address:
S P Khetan
Department of Microbiology, Government Medical College, Nagpur, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0255-0857.142248

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 ~ Abstract 

Cladophialophora bantiana brain abscess is a rare and frequently fatal infection, often seen in immunocompetent individuals. 34 year old immunocompetent woman who presented with convulsions is reported. She was initially treated with antituberculous drug. During 15 days of treatment, she deteriorated. Hence she underwent craniotomy, which revealed brain abscesses due to C. bantiana. Subsequently she was treated with fluconazole , but eventually succumbed to the infection on the 7th day of treatment. Mortality remains high with this rare mycosis, even in immunocompetent patients. The case illustrates the clinical and radiological similarities between tuberculoma and other etiologies of brain abscesses. This emphasizes the need to perform histological and microbiological studies prior to the initiation of any form of therapy.


Keywords: Cladophialophora bantiana, phaeohyphomycosis, brain abscess


How to cite this article:
Khetan S P, Agrawal V A, Qazi M S. Cerebral phaeohyphomycosis - could early diagnosis have saved the patient? . Indian J Med Microbiol 2014;32:440-2

How to cite this URL:
Khetan S P, Agrawal V A, Qazi M S. Cerebral phaeohyphomycosis - could early diagnosis have saved the patient? . Indian J Med Microbiol [serial online] 2014 [cited 2019 Dec 8];32:440-2. Available from: http://www.ijmm.org/text.asp?2014/32/4/440/142248



 ~ Introduction Top


Phaeohyphomycosis is a term used to describe infections caused by fungi that contain melanin in their cell walls. Cladophialophora bantiana is an uncommon, soil-based, phaeohyphomycotic fungus, which is neurotropic. [1] Clinical presentation includes haemiparesis, tonic spasm, headache, fever, sensory variation, cerebral irritation and even psychotic behavioural changes. [2] Combination of anti-fungal chemotherapy, surgical debridement and careful immunological interventions are strongly recommended to eradicate these intractable infections. [3] If untreated, it causes mostly fatal cerebral infections in both immunosuppressed and imunocompetent patients. [1]


 ~ Case Report Top


This case report describes the development of neurological symptoms in a 34-year-old postpartum female with delayed diagnosis and an adverse outcome.

Past history - She had delivered a baby boy by caesarean section and developed acute kidney injury during the postpartum period, for which she was referred to a tertiary care centre. She was put on haemodialysis through a double lumen venous catheter. She received haemodialysis for 2 weeks and subsequently her urine output improved, after which she was discharged from the hospital. She was re-admitted a week after discharge with generalised tonic-clonic convulsions, headache and vomiting.

Clinical examination - She was re-admitted in a drowsy state and was not able to move her four limbs. She had neck stiffness and rigidity. All vital parameter were within normal limits.

On examination of central nervous system (CNS), deep tendon reflexes were present in the upper limb and absent in the lower limb. Plantars were extensor. She also had bilateral crepitations on auscultation. Examination of other systems did not reveal any significant finding.

Investigations - All routine examinations  like complete blood cell count (CBC), liver function test (LFT) and kidney function test (KFT) were within the normal limits at the time of re-admission. Magnetic resonance imaging (MRI) scan of brain showed multiple confluent ring-shaped lesions in the right periventricular region of fronto-parietal area and genu of corpus callosum with significant perilesional oedema and mass effect over adjacent structures. There was compression of lateral ventricle, midbrain and 3 rd ventricle with midline shift. Ring lesions displayed central hypointense and peripheral mild hyperintense signals on T1 and central hyperintense and peripheral hypointense signal on T2 sequences with restricted diffusion. The findings were highly suggestive of multiple tuberculomas or abscesses with significant mass effect [Figure 1].
Figure 1: MRI image of the patient

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Initial treatment - Patient was put on anti-oedema measures, given a single dose of steroid and empirically on anti-tubercular drugs. During 15 days of treatment, her clinical condition kept on worsening, hence craniotomy was performed. Pus was aspirated from the lesions and surgical debridement of necrotic tissue was done. Pus was sent for microbiological examination and tissue for histopathological examination. Microbiological evaluation of the pus swab showed that the laboratory findings of Gram stained revealed pus cells, fungal hyphae but no bacteria. Ziehl Nielson method (20% H 2 SO 4 ) screening was negative for acid-fast bacilli.

Potassium hydroxide (KOH) mount showed numerous pigmented, septate hyphae. No bacterial growth was observed on culture.

Fungal cultures were done on Sabouraud dextrose agar (SDA) plain and SDA with actidione. The tubes were incubated at 25°C and 35°C. After 72 h of incubation, the tube showed colonies with an olive grey velvety appearance with a black undersurface [Figure 2]. For identification, slide culture was done which showed dark-walled septate hyphae with single-celled oval conidia in long-branched chains [Figure 3] and [Figure 4]. Hyphae were septate and darkly pigmented with many conidia attached to the sides and lying free. Conidia were one-celled, pale brown, smooth-walled and ellipsoid in shape. The conidiophores showed an acropetous type of branching. The isolate could be grown at 42°C and was also urease positive. Histopathological examination also showed a granulomatous inflammation along with numerous septate pigmented fungal hyphae. A diagnosis of phaeohyphomycosis was made based on the histopathological and mycological findings. The fungus was presumptively identified by its colony characteristic on SDA, morphology, urease production and its ability to grow at 42°C as a dematiaceous (black) fungus from C. bantiana hyphomycete species. Fungal growth was sent for confirmation and speciation to PGIMS, Chandigarh, and was identified as a rare fungus C. bantiana.
Figure 2: SDA with growth of C. bantiana incubated at room (R) and Incubator (I)

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Figure 3: Microscopic (40 ×) C. bantiana

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Figure 4: Lactophenol cotton blue mount of C. bantiana

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There are some conventional biochemical tests and physiological tests, which are commonly used in the identification of C. bantiana. This fungus grows at 42-43°C, it can grow on media that contains cycloheximide. It is urease positive and the Fontana-Masson staining stains it black. The Fontana-Masson stain is specific for melanin. This species is non-proteolytic on casein agar and it is unable to liquefy 12% gelatin. However, molecular methods like sequencing are the most dependable tools that can be used for its confirmation. [4]

Patient was further investigated for any immunocompromised condition. She was non-reactive for HIV 1 and II antibodies. Fungus was not isolated from any other sites like blood and sputum. Her blood sugar was found to be raised, although it had always been normal earlier with the possibility of steroid-induced high blood sugar, which could have contributed to her immunocompromised condition and deterioration.


 ~ Specific treatment and outcome Top


  Patient received Fluconazole 200 mg twice daily (BID) for 6 days after her craniotomy aspirated pus confirmed fungal aetiology. But, ultimately she succumbed on the 7 th day of anti-fungal therapy.


 ~ Discussion Top


C. bantiana is the most commonly isolated dematiaceous species from brain abscesses, although the portal of entry is not clearly established; most probably it is through respiratory tract. Inhalation of spores followed by haematogenous dissemination to brain is the likely possibility. [2] Other sources can be from paranasal sinus (allergic fungal sinusitis) or direct inoculation in brain. This condition is highly fatal and mortality rates are almost equal among immunocompromised and immunocompetent patients.

In this case, patient developed acute kidney disease (AKD) as a complication of postpartum period and underwent haemodialysis. She might have acquired fungal infection during her hospital stay. Our patient was a postpartum female with AKD, which could have been predisposing factors for making her susceptible for acquiring this uncommon fungal infection. Her immunity was further compromised due to steroid-induced high blood sugar.

Her MRI was suggestive of tuberculoma, cysticercosis or other causes of brain abscess. As tuberculosis (TB) is endemic in India, the patient was empirically put on anti-tubercular regime. She did not respond as it was a case of cerebral phaeohyphomycosis. As one of the differential diagnoses of MRI was suggestive of TB, the real diagnosis was delayed.

Patients with mass lesions in the brain should always have tissue sent for culture (including for fungi). Clinicians must consider fungal infection in the differential diagnosis of these patients, even if they are immunocompetent.

The patient died a week after receiving specific anti-fungal treatment. It is certain that the diagnosis was delayed as the patient was treated for a common endemic bacterial infection without even an iota of suspicion for an uncommon fungal infection. Even if the treatment had been initiated earlier, fungal brain abscess carries a poor prognosis, regardless of underlying disease. However, the case emphasises the need to perform histological and microbiological studies prior to the initiation of any form of therapy.

In cerebral phaeohyphomycosis cases, based on available data and experience, complete surgical resection appears to be necessary for optimal outcomes. Therapy with amphotericin B alone (standard or lipid preparations) may not be adequate. Some successfully treated cases used itraconazole and/or flucytosine in combination with amphotericin B, although few have been documented. The newer azole anti-fungal drugs, such as voriconazole and posaconazole, may also have a role in therapy, based on their good in vitro activity. [5] The optimal therapeutic regimen is not known, although polytherapy is better than monotherapy. Prolonged follow-up (>1 year) is essential because relapses are not uncommon. [6]


 ~ Acknowledgment Top


The authors are thankful to Dr. Arunalok Chakraborty, Professor, Department of Mycology, P. G. I., Chandigarh for confirmation of species of fungus.

 
 ~ 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.Borkar SA, Sharma MS, Rajpal G, Jain M, Xess I, Sharma BS. Brain abscess caused by Cladophialophora Bantiana in an immunocompetent host: Need for a novel cost-effective antifungal agent. Indian J Med Microbiol 2008;26:271-4.  Back to cited text no. 2
[PUBMED]  Medknow Journal  
3.Li DM, de Hoog GS. Cerebral phaeohyphomycosis: A cure at what lengths? Lancet Infect Dis 2009;9:376-83.  Back to cited text no. 3
    
4.Sutton DA, Rinaldi MG, Sanche SE. Dematiaceous fungi. In: Anaissie EJ, McGinnis MR, Pfaller MA, editors. Clinical Mycology. 2 nd ed. Philadelphia: Churchill Livingstone-Elsevier; 2009. p. 329 -54.  Back to cited text no. 4
    
5.Badali H, de Hoog GS, Curfs-Breuker I, Klaassen CH, Meis JF. Use of amplified fragment length polymorphism to identify 42 Cladophialophora strains related to cerebral phaeohyphomycosis with in vitro antifungal susceptibility. J Clin Microbiol 2010;48:2350-6.  Back to cited text no. 5
    
6.Lyons MK, Blair JE, Leslie KO. Successful treatment with voriconazole of fungal cerebral abscess due to Cladophialophora bantiana. Clin Neurol Neurosurg 2005;107:532-4.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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