Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 9  |  Issue : 4  |  Page : 518-521  

An unusual case of invasive rhinocerebral aspergillosis in an immunocompetent woman


Department of Microbiology, Institute of Postgraduate Medical Education and Research, Kolkata, West Bengal, India

Date of Web Publication12-Jul-2016

Correspondence Address:
Kalidas Rit
70B, T.C. Mukherjee Street, P.O. Rishra, Hooghly - 712 248, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-2870.186075

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  Abstract 


Invasive rhinocerebral aspergillosis is very rare and commonly seen in immunocompromised individuals with a high rate of morbidity and mortality. It is an opportunistic infection and has a spectrum of presenting features from allergic sinusitis to active invasive disease. Here, we describe a case of invasive rhinocerebral aspergillosis in an immnunocompetent middle-aged woman. She was treated with systemic antifungal medications, but she ultimately succumbed to death.

Keywords: Immunocompetent, invasive, rhinocerebral aspergillosis


How to cite this article:
Pal N, Rit K. An unusual case of invasive rhinocerebral aspergillosis in an immunocompetent woman. Med J DY Patil Univ 2016;9:518-21

How to cite this URL:
Pal N, Rit K. An unusual case of invasive rhinocerebral aspergillosis in an immunocompetent woman. Med J DY Patil Univ [serial online] 2016 [cited 2022 Oct 5];9:518-21. Available from: https://www.mjdrdypu.org/text.asp?2016/9/4/518/186075




  Introduction Top


Aspergillus is ubiquitous fungus that lives in soil and decaying vegetation. The incidence of invasive aspergillosis is increasing over the last few years and usually associated with some form of immunodeficiency.[1] Primary sites of involvement are paranasal sinuses in immunocompetent individuals and lungs in immunocompromised individuals.[2] The manifestation of fungal diseases of paranasal sinuses ranges from allergic sinusitis to active invasive disease. Involvement of central nervous system (CNS) in invasive aspergillosis is usually associated with nearly 100% mortality. CNS aspergillosis in immunocompetent individuals has been considered an uncommon disease responsible for <5% of all CNS infections.[3] Milosev in 1966 first reported the case of invasive aspergillosis of the paranasal sinuses,[4] followed by similar reports from other tropical countries like India.[2],[3] Here, we report a rare case of invasive rhinocerebral aspergillosis in an immunocompetent individual.


  Case Report Top


A 45-year-old woman presented with complaints of numbness and accumulation of food on the right side of the mouth for the last 5 months along with diplopia in the right eye and vertigo for the last 1-month. The patient had history of small of amount of nasal bleeding 5 months ago and right-sided headache for last 3 months. There was no history of fever and convulsions. She was treated by local doctors in Jamshedpur. There was no history of diabetes, hypertension, tuberculosis, and asthma. On admission, the patient was conscious, oriented with pulse rate of 94/min and blood pressure of 130/80. The patient was thin built and moderate pallor was present. No icterus, clubbing, cyanosis, edema, and lymphadenopathy were found. There was difficulty in opening of mouth along with fifth and sixth cranial nerves paresis. Other systemic examinations did not reveal any abnormal findings. Routine blood examination report revealed Hb was 7.6 g%, total leucocyte count 8200 with neutrophils 80%, lymphocytes 12%, monocytes 2%, eosinophils 6%, and fasting blood sugar was 90 g/dl. Serological tests for HIV antibodies, HbsAg, and hepatitis C virus antibodies were all negative. Her chest X-ray was normal. Magnetic resonance imaging (MRI) of the brain revealed the large areas of intense patchy enhancing infiltrating destructive lesion in the right basifrontal region predominantly involving the ethmoidal sinuses, nasal cavity, medial wall of the right orbit, and right maxillary antrum [Figure 1]. Subsequently, the patient developed proptosis of the right eye. Histopathological examination of collected specimen from the right maxillary antrum showed numerous acute angled branching septate hyaline hyphae [Figure 2]. Oral itraconazole (200 mg/day) and intravenous (IV) amphotericin B were started and continued for 2 weeks. However, the patient's clinical conditions further worsened; she developed left-sided paraplegia along with the right eye proptosis and severe headache. Enucleation of the right eye was done, and retro orbital mass was sent for fungal culture and histopathological examination. Direct KOH microscopy of collected specimen showed plenty of acute angled branching septate hyphae [Figure 3]. On Sabouraud's Dextrose Chloramphenicol Agar (SDCA) media greenish growth of Aspergillus fumigatus was demonstrated after 7 days of incubation at 22°C [Figure 4]. Lactophenol cotton blue preparation of growth on SDCA media showed typical conidiophores with conidia of A. fumigatus [Figure 5]. The patient was switched over to IV voriconazole and echinocandin therapy. However, the conditions worsened further and patient ultimately succumbed to death.
Figure 1: Magnetic resonance imaging brain of affected individual showing infiltrating patchy parenchymal lesion

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Figure 2: Histopathological examination of collected specimen from right maxillary antrum

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Figure 3: KOH mount preparation showing plenty of acute angled branching septate hyphae

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Figure 4: Growth of Aspergillus fumigatus on Sabouraud's Dextrose Chloramphenicol Agar media

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Figure 5: Lactophenol cotton blue preparation from growth showing typical conidiophores with conidia of Aspergillus fumigatus

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  Discussion Top


Invasive aspergillosis is usually a disease of immunocompromised individuals often associated with rapid progression and fatal outcome. Various factors such as profound neutropenia, bone marrow transplantation, terminal HIV infection, and chronic granulomatous disease often predispose to invasive aspergillosis.[5] Invasive aspergillosis is characterized by invasion of lymphatics and vasculature with multifocal involvement. This form of disease in immunocompetent individuals has been described in literature,[4] but very few reports have been published from this country.[2] Our patient developed invasive rhinocerebral aspergillosis without any underlying immunodeficiency in the form of diabetes, alcoholism, past respiratory infections, and other immunodeficiency diseases. It has been reported that frequencies of aspergillosis are high in tropical and subtropical regions of Asia, Africa, South and Central America where fungus thrive well due to hot and humid climate. These countries have a large number of poor people with few modern medical facilities which led to an increase in the number of invasive fungal diseases with high mortality. The misuse of steroids and intravenous drugs, availability of spurious medical care infusion sets, and untrained health care providers are possible additional risk factors of invasive aspergillosis in apparent immunocompetent individuals in those countries. Natural immunity such as normal anatomical barriers, humoral factors such as complement, phagocytic cells, and cell-mediated immunity plays a major role in the defense against invasive aspergillosis. A subtle impairment of phagocytic functions or T-cell activity which we were unable to demonstrate may be responsible for primary invasive aspergillosis. Aspergillosis of paranasal sinuses is rare, but the number of reported cases is increasing globally. Infection is initially confined to a single sinus, and usually, maxillary is the most commonly involved sinus followed by sphenoid. The clinical manifestations are more prominent in persons having underlying immunodeficiency. The clinical presentation is usually nonspecific, and the disease condition is often asymptomatic in immunocompetent individuals resulting in delayed diagnosis. Aspergillosis of CNS is rare, involving mainly immune suppressed individual and accounts for only 5% of intracranial fungal infections.[3] The infection may spread via two ways either by hematogenous route or by direct extension from an area anatomically adjacent to the brain. Neuroaspergillosis may manifest in several forms such as persistent meningitis, encephalitis, meningoencephalitis, mycotic, ischemic, hemorrhagic infarcts, stroke-like syndrome, intracranial space occupying lesion, skull base syndrome, brain abscess, intraorbital space occupying lesion, and aspergilloma.[6] Very few case reports of neuroaspergillosis in immunocompetent individuals were published from India, one by Murthy et al. from Hyderabad in 2001,[2] and another by Sood et al. from Rajasthan in 2007.[7] Our patient was apparently immunocompetent, and no underlying risk factors were present. Probably, in this case, intracranial spread occurred by erosion of the base of the skull and along with the blood vessels. Computed tomography is helpful to know the magnitude of bony involvement. MRI is usually advised for meningeal or intraparenchymal involvement. Conventional direct microscopy, histopathology and culture methods are the only available diagnostic techniques in most centers of developing countries to diagnose invasive aspergillosis due to nonavailability of galactomannan or beta-glucan tests assay. In our case diagnosis was done by histopathological examination, direct KOH microscopy, and positive culture report. Speciation of A. fumigatus was made by demonstrating characteristic grayish green colony and typical conidiophores with flask-shaped vesicle producing single series of phialides only on the upper half of vesicles. Regarding the therapeutic option, the commonly recommended drug is voriconazole with a success rate of nearly 53% followed by amphotericin-B with success rate 32%.[8],[9] Regarding the other therapeutic options itraconazole, caspofungin and posaconazole are useful. Itraconazole is useful in patient with less immunosuppression.[9] Our patient received initial combination therapy of itraconazole with amphotericin-B followed by combination therapy of voriconazole and echinocandin. However, the patient ultimately succumbed to death in spite of aggressive therapeutic approach.


  Conclusion Top


We can say that high index of clinical suspicion in an apparently immunocompetent patient is necessary for early diagnosis and aggressive surgical management along with antifungal medications offers the best hope for survival.

Acknowledgments

Professor Prasanta Kumar Maiti, Professor and Head, Department of Microbiology, Institute of Postgraduate Medical Education and Research, 244 AJC Bose Road, Kolkata - 700 020, West Bengal, India.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Bennett JE. Aspergillosis. In: Isselbacher KJ, Braunwald E, Wilson JD, editors. Harrison's Principles of Internal Medicine. 13th ed. New York: McGraw Hill; 1995. p. 855, 861-2.  Back to cited text no. 1
    
2.
Murthy JM, Sundaram C, Prasad VS, Purohit AK, Rammurti S, Laxmi V. Sinocranial aspergillosis: A form of central nervous system aspergillosis in South India. Mycoses 2001;44:141-5.  Back to cited text no. 2
    
3.
Karim M, Alam M, Shah AA, Ahmed R, Sheikh H. Chronic invasive aspergillosis in apparently immunocompetent hosts. Clin Infect Dis 1997;24:723-33.  Back to cited text no. 3
    
4.
Miloshev B, Davidson CM, Gentles JC, Sandison AT. Aspergilloma of paranasal sinuses and orbit in Northern Sudanese. Lancet 1966;1:746-7.  Back to cited text no. 4
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5.
Samir M, Masoud AR. Sinus aspergillosis and allergic fungal sinusitis. Ann Saudi Med 1996;16:395-9.  Back to cited text no. 5
    
6.
Kleinschmidt-DeMasters BK. Central nervous system aspergillosis: A 20-year retrospective series. Hum Pathol 2002;33:116-24.  Back to cited text no. 6
    
7.
Sood S, Sharma R, Gupta S, Pathak D, Rishi S. Neuroaspergillosis in an immunocompetent patient. Indian J Med Microbiol 2007;25:67-9.  Back to cited text no. 7
[PUBMED]  Medknow Journal  
8.
Schwartz S, Thiel E. CNS-aspergillosis: Are there new treatment options? Mycoses 2003;46 Suppl 2:8-14.  Back to cited text no. 8
    
9.
Schwartz S, Ruhnke M, Ribaud P, Corey L, Driscoll T, Cornely OA, et al. Improved outcome in central nervous system aspergillosis, using voriconazole treatment. Blood 2005;106:2641-5.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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