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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 54  |  Issue : 3  |  Page : 111-113

A diagnostic challenge of invasive sellar neuroaspergillosis in an immunocompetent patient


1 Department of Pathology, Institute of Medical Sciences and SUM Hospital, Bhubaneswar, Odisha, India
2 Department of Neurosurgery, Institute of Medical Sciences and SUM Hospital, Bhubaneswar, Odisha, India

Date of Submission19-Oct-2020
Date of Decision24-Nov-2020
Date of Acceptance27-Jan-2021
Date of Web Publication12-Jun-2021

Correspondence Address:
Pranita Mohanty
Department of Pathology, Institute of Medical Sciences and SUM Hospital, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/fjs.fjs_194_20

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  Abstract 


The commonest differential for sellar space occupying lesion is tumor, and clinicians would rarely consider aspergillosis at this location in an immunocompetent patient. Hence, a high index of suspicion clinically and histological confirmation are required to reach the diagnosis. Here, we report a case of such, a 28-year-old immunocompetent male presented with headache, vomiting, and diplopia for 7 days without any history of convulsion, unconsciousness or nasal symptoms. His magnetic resonance imaging and computed tomography of the brain and pituitary gland were suggestive of a primary bone tumor or pituitary macroadenoma of the sellar region. He then underwent surgery, and intraoperative squash cytology, frozen section, and fine-needle aspiration cytology of aspirated pus revealed necrotizing granulomatous lesion of fungal etiology. Postoperative histopathology and special stains (periodic acid-Schiff, Gomori methenamine silver) confirmed invasive aspergillosis, and Aspergillus flavus was isolated by pus culture. The surgery was followed by systemic voriconazole therapy, and there were no further complications.

Keywords: Aspergillus flavus, invesive aspergillosis, sellar region


How to cite this article:
Mohanty P, Lenka A, Govardhan T, Panigrahi S. A diagnostic challenge of invasive sellar neuroaspergillosis in an immunocompetent patient. Formos J Surg 2021;54:111-3

How to cite this URL:
Mohanty P, Lenka A, Govardhan T, Panigrahi S. A diagnostic challenge of invasive sellar neuroaspergillosis in an immunocompetent patient. Formos J Surg [serial online] 2021 [cited 2021 Jul 24];54:111-3. Available from: https://www.e-fjs.org/text.asp?2021/54/3/111/318213




  Introduction Top


CNS aspergillosis (Neuroaspergillosis) in immunocompetent patients is relatively rare, usually manifests as isolated brain lesions, and is frequently misdiagnosed and undertreated. Only a few dozen cases (34 cases) have been described to date, of which sellar location is described in only one.[1] Aspergillosis is caused by ubiquitous saprophytic fungus of Aspergillus species. The commonest being Aspergillus fumigatus, but Aspergillus flavus and Aspergillus niger can also be rarely seen.[2],[3]

Most cases of neuroaspergillosis due to A. flavus have been reported from India, Pakistan, the Middle East and Africa.[4] A flavus is more virulent and resistant to common antifungal drugs. Two modes of entry exists; (1) Through airborne conidia into the respiratory tract and paranasal sinuses (PNS) causing granuloma in the paranasal sinus with direct spread or through focal bone destruction, or through tiny foramina or venous nexus even without bone destruction into central nervous system (CNS) in immunocompetent patients.


  Case Report Top


A 28-year-old immunocompetent, nondiabetic, hypertensive male presented to neurosurgery department with complaints of headache, vomiting, and diplopia for 7 days. He had no history of convulsion, loss of consciousness, sign of meningism, nasal symptoms, or immunosuppressive conditions such as cancer or autoimmune disease, or drug induced/trauma/surgery associated conditions. The patient was conscious, oriented, afebrile, and had Glasgow Coma Scale-15/15, normal visual acuity, B/L plantar flexor, but VI cranial nerve palsy (left > right) was noted. Routine hematological investigation showed total leukocyte count-18.54 × 103/cmm, neutrophilic leukocytosis with differential leukocyte count: (N-85, E-01, L-10, M04), Triple H (HIV, HBsAG, HCV) test – negative, erythrocyte sedimentation rate-16 mm/1st h. His computed tomography (CT) angiography of the brain with PNS showed poorly circumscribed hypo-enhanced soft-tissue mass lesion involving skull base with clival erosion. Cavernosal segment of bilateral internal carotid artery showed 180° encasement with complete involvement of bilateral cavernous sinuses. Giant cell tumor and chordoma were possibilities. Subsequent magnetic resonance imaging (MRI) of the brain and pituitary gland revealed an enhanced mass lesion with the epicenter in clivus and sphenoidal sinus measuring 41 mm × 47 mm × 36 mm, and old ischemic changes in bilateral cerebral white matter were seen [Figure 1]a. Findings were suggestive of primary bone neoplasm or invasive pituitary macro adenoma [Figure 1]a, [Figure 1]b, [Figure 1]c. The mass did not involve the orbit by MRI. The endoscopic surgery was done by transsphenoidal approach. An encapsulated friable solid mass with a cyst was encountered which had eroded the sphenoid bone and sellar base. About 4 ml of frank pus was expelled out from the cystic part, which was sent for both cytology and culture study. Then the solid friable tissue was dissected out and sent for squash cytology, frozen section, and histopathology.
Figure 1: (a) Magnetic resonance imaging showing large expansile lesion in sellar region, (b and c) bilateral encasement of ICA, (d and e) Squash cytology showing many multinucleated giant cells engulfing fungal hyphae (inset with red arrow), and epithelioid cell clusters, (×40; DIFF-QUIK, h and e Stain)

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Pus and squash cytology [Figure 1]d and [Figure 1]e revealed mixed inflammatory cells such as histiocytes and cyst macrophages, and a few epithelioid cell clusters along with many multinucleated giant cells engulfing fungal elements of both hyphal and conidial forms in a necrotic background. These findings suggested that the lesion was necrotizing granuloma of fungal etiology in the sellar region.

Frozen section and postoperative histopathology [Figure 2]a and [Figure 2]b showed dense mixed inflammatory cell infiltration, including polymorphs, lymphocytes, plasma cell, histiocytes, occasional epithelioid cell cluster, and many multinucleated giant cells engulfing fungal elements embedded in the brain parenchyma with hemorrhage and necrosis. The fungi showed broad septate branching hyphae and occasional round or oval budding forms. Histopathological diagnosis was compatible with Fungal Granuloma, sellar region (Invasive Aspergillosis). Gomori methenamine silver and periodic acid-Schiff special stain identified the fungal elements as Aspergilus strain [Figure 2]c and [Figure 2]d. Pus culture revealed Aspergillus flavus species. He was treated with Fluconazole (150 mg) 1 tab once a day × 15 days beginning on the 4th postoperative day, followed by Voriconazole (200 mg) 1 tab twice a day for 15 days. MRI was repeated on the 7th and 20th day postoperation and the patient was examined at 6 months. The patient was free of all symptoms and without any residual lesion. No further complication or new neurological deficit was detected.
Figure 2: (a and b) Photomicrograph showing fungal granuloma with many multinucleated giant cells, some engulfing fungal hyphae (red arrow) (100x&40x; H and E stain), (c and d) Special stains (40×; Gomori methenamine silver and periodic acid–Schiff respectively) highlighting the fungal hyphae (red arrow) and conidia (blue arrow)

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  Discussion Top


A total of 34 neuroaspergilosis cases in immunocompetent patients have been described to date, mostly as case reports except one case series; of which sellar location was observed in one case only.[1] They usually present with clinical manifestations of headache, visual impairment, diplopia, hemiplegia, fever, and epilepsy similar to that of a tumor. Possible causes/risk factors are: hot and dry Indian environment with abundance of Aspergillus spores, trauma, previous surgery, malnutrition, and use of steroids or anti-tubercular drugs. Aspergillus flavus is the most common organism to cause diverse clinical spectrum broadly categorized into four types: (1) Invasive aspergillosis, (2) Subacute/chronic infections, (3) Allergic infections, and (4) Posttraumatic infections. Except the fourth category, in all other types the most common organs involved is lungs, followed by PNS and CNS. The lesions in neuroaspergillosis are usually located in cerebrum, intracranial-extradura, or invading orbit and skull base, whereas sellar location is very rare. The term Invasive Aspergillosis is used where the fungal hyphal forms are found histologically within the tissues as in our case. Sometimes, the cause is unclear as in the present case; he neither had any nasal symptoms nor specific immunosuppressive condition, trauma, contact history, or neurosurgical procedure. Although an enhanced mass lesion (annular or nodular) and involvement of major vessels and cavernous sinus with ICA encasement is a frequent feature in MRI as in our case, it is not specific and can be seen in many tumors, hence that poses a radiologic diagnostic challenge.[5] The use of different modalities of investigation such as MRI, CT angiography, cerebrospinal fluid (CSF) routine study, CSF culture, and next-generation sequencing test can facilitate early diagnosis, whereas histopathology exam of surgical sample supplemented with special stain and culture is confirmatory. Surgical resection allows a histopathological diagnosis. Voriconazole is the new gold standard drug and Amphotericin B should be avoided.[6]

Amphotericin B should be avoided.[6] Literatures show that voriconazole alone is effective in most cases, while a combination antifungal therapy of-fluconazole/amphotericin B/Voriconazole/isavuconazole/Itraconazole is also advisable. The outcome is usually encouraging (100%–69%) as it is a benign and curable disease.[7]


  Conclusion Top


Correct diagnosis of sellar aspergillosis can only be achieved by histopathology. Surgical intervention with antifungal treatment is optimal for this curable disease to save the life of patient like in the present case.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initial will not be published and due efforts will be made to conceal the identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Ma Y, Li W, Ao R, Lan X, Li Y, Zhang J, et al. Central nervous system aspergillosis in immunocompetent patients: Case series and literature review. Medicine (Baltimore) 2020;99: e22911.  Back to cited text no. 1
    
2.
Phuttharak W, Hesselink JR, Wixom C. MR features of cerebral aspergillosis in an immunocompetent patient: Correlation with histology and elemental analysis. AJNR Am J Neuroradiol 2005;26:835-8.  Back to cited text no. 2
    
3.
Kumar D, Nepal P, Singh S, Ramanathan S, Khanna M, Sheoran R, et al. CNS aspergilloma mimicking tumors: Review of CNS aspergillus infection imaging characteristics in the immunocompetent population. J Neuroradiol 2018;45:169-76. https://doi.org/10.1016/j.neurad.2017.11.001.  Back to cited text no. 3
    
4.
Hedayati MT, Pasqualotto AC, Warn PA, Bowyer P, Denning DW. Aspergillus flavus: Human pathogen, allergen and mycotoxin producer. Microbiology 2007;153:1677-92.  Back to cited text no. 4
    
5.
Marzolf G, Sabou M, Lannes B, Cotton F, Meyronet D, Galanaud D, et al. Magnetic resonance imaging of cerebral aspergillosis: Imaging and pathological correlations. PLoS One 2016;11: e0152475.  Back to cited text no. 5
    
6.
Rudramurthy SM, Paul RA, Chakrabarti A, Mouton JW and Meis JF. “Invasive Aspergillosis by Aspergillus flavus: Epidemiology, Diagnosis, Antifungal Resistance, and Management.” J Fungi 2019;5:55.1-23. doi:10.3390/jof5030055.  Back to cited text no. 6
    
7.
Candoni A, Klimko N, Busca A, Di Blasi R, Shadrivova O, Cesaro S, et al. Fungal infections of the central nervous system and paranasal sinuses in onco-haematologic patients. Epidemiological study reporting the diagnostic-therapeutic approach and outcome in 89 cases. Mycoses 2019;62:252-60.  Back to cited text no. 7
    


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