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CASE REPORT |
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| Year : 2014 | Volume
: 9
| Issue : 2 | Page : 159-161 |
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Multiple intracranial fungal abscesses in an immunocompetent infant treated surgically
Ashutosh Khandelwal, Noufal Basheer, Ashok K Mahapatra
Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi, India
| Date of Web Publication | 21-Aug-2014 |
Correspondence Address:
Ashok K Mahapatra
Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi - 110 029
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/1817-1745.139332
 Abstract | | |
Intracerebral abscess are rare in neonates and infants. We present a rare case of 4-month-old immunocompetent infant with multiple intracranial fungal abscess. Right frontoparietal craniotomy was done with excision of multicystic mass containing 200cc of dirty colored fluid. Biopsy and culture of abscess came as Aspergillus fumigates. Patient received parental amphotericin B. An immunocompetent infant with intracranial aspergillosis is rare. We advocate combination of surgical excision with parental amphotericin B as best way to achieve good result.
Keywords: Amphotericin B, , fungal abscess, infant, multiple
How to cite this article:
Khandelwal A, Basheer N, Mahapatra AK. Multiple intracranial fungal abscesses in an immunocompetent infant treated surgically. J Pediatr Neurosci 2014;9:159-61 |
| Introduction | |  |
There are over 350 recognized species of Aspergillus, only few are pathogenic to man. Invasive aspergillosis predominantly occurs in immunocompromised hosts, such as those with hematological malignancies, hematopoietic stem cell or solid organ transplants, congenital or acquired immunodeficiency, as well as prolonged use of corticosteroids and other immunosuppressive drugs. [1] Other situations that may predispose to invasive aspergillosis infections include intravascular catheters, premature birth, severe burns, abdominal surgery, and intravenous drug addiction. [2],[3]
Dissemination of aspergillosis is relatively common, with the central nervous system (CNS) being one of the most frequent sites of invasive aspergillosis after the lungs. [4] The brain and subarachnoid space are protected from fungal pathogens by anatomic and functional barriers, but under special conditions and immune system abnormalities, fungal pathogens can breach these barriers. CNS symptoms eventually develop in one-half of patients with disseminated aspergillosis and are the presenting feature in one-third of them. [5] The extent of gross neuropathologic disease ranges from subtle abscesses, focal purulent meningitis and bland infarctions to massive hemorrhagic necrosis causing herniation and death. Despite perceived advances in early diagnosis, initiation of treatment and newer antifungal agents, CNS aspergillosis remains a devastating opportunistic infection and its prognosis is discouragingly poor even in adults [6] and more so in infants. We present a rare case of a 4-month-old immunocompetent infant with intracranial fungal abscess.
| Case Report | | |
A 2-month-old infant presented to a local hospital with high fever for 4 weeks and was investigated for the fever by complete blood count with microscopy for malarial parasite as its endemic in Asian population which came out to be negative. X-ray chest was done to rule out lung infection, blood cultures were negative for bacteremia. Lumbar cerebro spinal fluid was nonmeningitic. Two-dimensional echocardiography was done which was also normal. The patient was referred to our hospital at the age of 4 months with persistent moderate degree fever for 12 weeks. General physical examination was normal with no focus of infection and his chest examination was normal. Patient weighed 4 kg. Head circumference was within normal limits with tense anterior fontanelle. The child was fixing gaze and social smile was present. Child was moving all the 4 limbs however there was mild left hemiparesis. He was started on empiric antibiotics. Mantoux test and viral marker were negative. IgA and IgG were also normal. Magnetic resonance imaging showed heterointense ill-defined mass lesion in right fronto-temporal-parietal area with mild peripheral, septal contrast enhancement, perilesional edema with mass effect and midline shift [Figure 1]. Differential diagnosis was malignant intracranial tumor like primitive neuroectodermal tumor or focal cerebritis/encephalitis. | Figure 1: Contrast-enhanced magnetic resonance imaging sagittal view showing heterointense ill-defined mass lesion in right fronto-temporal-parietal area with mild peripheral edema
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As the fever was not responding and no cause of fever was identified, primary intracerebral infective pathology was also strongly considered and right frontoparietal craniotomy with excision of multicystic mass containing 200 cc of dirty colored fluid done. The wall of the cyst was sent for various cultures and histopathology. Fungal smear with culture came out to be Aspergillus fumigatus and histopathology showed branching hyphae consistent with fungal abscess [Figure 2]. Amphotericin B was started at 1 mg/day intravenously, and was gradually escalated to the daily dose of 0.7-1 mg/kg/day. The total dose of 250 mg was administered over a period of 6 weeks. His fever subsided following treatment and was discharged. Follow-up contrast-enhanced computed tomography showed postoperative changes and no midline shift. At follow-up after 6 months, patient was afebrile and hemiparesis had improved. | Figure 2: Sections examined showed numerous granulomas with foreign-body type of giant cells. In one of the giant cells a pigmented septate hypha was seen. The morphological features were those of a aspergillosis [H and E, ×20]
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| Discussion | | |
Intracranial aspergillosis is a very rare condition in infants and neonates. The mortality rate in CNS aspergillosis is exceeding 90% and even approaching 100% in adult patients with acute leukemia with or without bone marrow transplantation. The mortality is no lower than approximately 70% even in the recent published study after administration of voriconazole with or without surgery. [1],[6],[7]
About 14 cases of intracranial aspergillosis in infants have been reported till recently [8],[9],[10],[11] among them only 2 had multiple brain abscess. Four of the 14 cases were operated of whom only 3 survived. Mortality rates were as high as 70% in surgically treated patients and in patient who was managed conservatively mortality was close to 100% with only one infant surviving.
Aspergillus species can induce unusual clinical features caused by unique sites of CNS involvement. Specifically, basilar meningitis, myelitis, sino-orbital disease, carotid artery invasion, epidural abscesses, massive hemorrhagic necrosis with herniation as the cause of death, isolated or subtle intracranial disease, and mycotic aneurysms can all be caused by Aspergillus spp. In addition, hemorrhagic necrosis as the primary macroscopic feature of CNS aspergillosis has been emphasized in some studies. [12],[13] In another review, single or multiple brain abscesses were the most common type of CNS aspergillosis and were found in 45 of 65 (69.2%) cases. [14] In our case also there were multiloculated brain abscesses.
Aspergillus species are primarily respiratory pathogens, and the lungs constitute the main portal of entry prior to dissemination. It is important to note that in most cases of CNS aspergillosis the portal of entry and the route for Aspergillus species remains unspecified and is a subject of speculation. In a few cases, CNS was the primary site of aspergillosis.
Underlying diseases seems to be a very important factor for the development of CNS aspergillosis. Prematurity, including transplacental inoculation [15],[16] were a risk factor for neonatal intracerebral aspergillosis suggesting that immature neonatal anatomical and immune defense systems probably play a role in disease acquisition. Prematurity, chronic granulomatous disease and a complex of diarrhea, dehydration, malnutrition, and invasive bacterial infections accounted for the majority of underlying conditions.
Invasive aspergillosis is exceedingly rare among individuals with apparently normal host defenses. In a 10-year retrospective analysis from the Hospital for Sick Children in Toronto, Canada, only one of 39 children and adolescents with definite or probable invasive aspergillosis was found to be immunocompetent. [17] The primary risk factors for invasive aspergillosis are prolonged and profound neutropenia and administration of high doses of corticosteroids. [18],[19] Not surprisingly, the cumulative frequency of invasive aspergillosis in patients with chronic granulomatous disease is exceedingly high and ranges from 16% to 40%. [20],[21]
Amphotericin B has been the mainstay of medical treatment for both suspected and proven CNS aspergillosis in childhood during the last decades. We consider combination of complete resection with parental amphotericin as a right strategy to achieve good results.
| Conclusion | | |
An immunocompetent infant with right fronto-temporo-parietal aspergillosis is rare and with good postoperative result is exceedingly rare. Preoperative diagnosis on basis of imaging or cerebrospinal fluid culture is difficult. Pathogenesis is uncertain while transplacental route remains as one possibility in our case. We recommend surgical resection with postoperative parental amphotericin as best therapy at present.
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[Figure 1], [Figure 2]
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