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|LETTER TO EDITOR
|Year : 2015 | Volume
| Issue : 3 | Page : 439-441
"Fungimitosis": Invasive fulminant Aspergillus brain infection mimicking gliomatosis cerebri
Alugolu Rajesh1, Srikant Reddy1, Malladi Venkat Surya Subbalaxmi2, Megha Shantveer Uppin3
1 Department of Neurosurgery, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Telangana, India
2 Department of Medicine, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Telangana, India
3 Department of Pathology, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Telangana, India
|Date of Web Publication||5-Jun-2015|
Department of Neurosurgery, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Telangana
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Rajesh A, Reddy S, Subbalaxmi MV, Uppin MS. "Fungimitosis": Invasive fulminant Aspergillus brain infection mimicking gliomatosis cerebri. Neurol India 2015;63:439-41
|How to cite this URL:|
Rajesh A, Reddy S, Subbalaxmi MV, Uppin MS. "Fungimitosis": Invasive fulminant Aspergillus brain infection mimicking gliomatosis cerebri. Neurol India [serial online] 2015 [cited 2019 Sep 17];63:439-41. Available from: http://www.neurologyindia.com/text.asp?2015/63/3/439/158250
Fungi are commensals in humans.  However, whenever host defenses are at stake, fungi can lead to opportunistic infections, some of which can be lethal. Aspergillosis is an infection caused by fungi of the Aspergillus fumigatus family. Commonly, maxillary sinusitis of dental origin and pulmonary aspergillosis act as primary sources of the infection.  Central nervous system (CNS) aspergillosis can affect the cerebral parenchyma, the meninges or the vascular system. Rarely, the infection may also be air-borne, contaminating the operative field during a neurosurgical procedure. 
A 50-year-old male patient presented with gradually progressive, holocranial, and dull aching headache for 4 months, associated with memory deficits over a period of 3 months. He had mild clumsiness in using his right hand. There was neither any history of co-morbid conditions nor any evident features suggestive of an immunocompromised state. On examination, the patient had a mini-mental state examination score of 24/30. His motor system examination was unremarkable except for positive pronator drift on the right side along with ipsilateral upgoing plantars. His hematological investigations were normal including nonreactivity for human immunodeficiency virus (HIV).
Cranial magnetic resonance imaging showed a diffuse lesion, hypointense on T1WI and hyperintense on T2WI, involving the left frontal, parietal and temporal lobes with patchy contrast enhancement. There was diffuse hemispheric edema of the same lobes with mass effect and midline shift [Figure 1]a-f. These features were suggestive of gliomatosis cerebri. Hence, the patient underwent a left fronto-temporo-parietal (FTP) craniotomy, left temporal lobectomy with decompression, and biopsy of the lesion from the frontal lobe via the middle frontal gyrus approach. At surgery, the lesion was found to be grayish white, minimally vascular, firm to hard (gritty) in consistency, which raised the suspicion of an infective etiology. Hence, microbiological staining (Gram's, KOH preparation, acid-fast bacillus staining) and cultures (aerobic, tubercular, and fungal) of the sample were sought, apart from its histopathogical examination. In the immediate postoperative period, the patient developed aphasia and right hemiplegia although this improved gradually over a period of 4 days. The histopathological examination showed fungal hyphae in the excised tissue and culture of the tissue suggested the diagnosis of aspergillus infection. The patient was started on intravenous amphotericin-B on postoperative day 2 (dose 1.5 mg/kg wt/daily). During the course in the hospital, the patient deteriorated in sensorium, and a repeat computed tomography (CT) of the brain showed diffuse cerebral edema with mass effect for which a left FTP decompressive craniectomy was performed. Over the next 2 days, the patient did not show any improvement in the neurological status, prompting us to repeat his CT head. The CT scan showed dilated ventricles for which an external ventricular drainage was instituted. The patient, however, continued to deteriorate and finally succumbed to the illness on the 8 th postoperative day.
|Figure 1: Magnetic resonance imaging of the brain showing a lesion which is hypointense on T1W images (a) and hyperintense on T2W and fluid attenuation inversion recovery images (b and c) involving the left frontal, temporal, and parietal regions with patchy enhancement on contrast imaging (d, e, f)|
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Invasive fungal infection involving the central nervous system (CNS) is often fulminant and fatal with a reported mortality of nearly 100% in the older literature. This disease was described as "almost always a clinical surprise" by Nadkarni and Goel.  These cases present an interesting diagnostic dilemma for the neurosurgeons and neuroradiologists, as neither the clinical nor the imaging features of CNS fungal infections are truly diagnostic of the disease and with the patient being immuno-competent, the possibility of fungal infection is seldom considered in the list of clinical differentials.  Cerebral parenchymal lesions can range from fungal granuloma, diffuse cerebritis to frank abscess formation. A sinonasal disease with intracranial extension is the most frequent presentation although isolated parenchymal lesions without evidence of any sinonasal involvement are not infrequent. They usually present as an heterogenous signal intensity lesion on T1WI and T2WI images, often showing areas of diffusion restriction within. Because of rarity of the lesion and its non-specific imaging findings, most cases are often confused with tumors and infarcts. In our case, involvement of one whole hemisphere mimicked gliomatosis cerebri; hence, we named it as "fungimitosis." Impaired/poor nutritional status, injudicious use of steroids or antituberculous drugs, and parasitic infections are all considered as predisposing factors for fungal infections. ,
Though no specific guidelines are available, early aggressive surgery followed by aggressive empirical antifungal therapy appears to be a reasonable approach considering the remarkable invasiveness of Aspergillus and its potential complications.  The patients with CNS Aspergillosis often show a peculiar response to surgery characterized by a disease flare up which may at times be fulminant and refractory to the medical management. The reason for this unique stormy postoperative course is incompletely understood but immunocompromise secondary to surgery and/or the stimulation of previously quiescent fungi due to surgical tissue handling have been proposed as the offending factors. , Aspergillus infection may uncommonly cause cerebral vasculitis leading to thrombosis of vessels and large infarcts which may contribute to the poor outcome after surgery.  Hence, it is not surprising that the prognosis of CNS aspergillosis is mostly unfavourable
To conclude, CNS Aspergillus infection, although rare, can affect immunocompetent individuals. The protean clinical manifestations as well as diffuse brain parenchymal involvement may lead to a diagnostic dilemma. Although the prognosis remains largely poor, an early diagnosis and radical surgical management followed by aggressive antifungal chemotherapy may help to improve the outcome.
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