- Department of Histopathology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
- Department of Neurosurgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
- Department of Endocrinology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
- Department of Radiology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
Correspondence Address:
Kanchan K. Mukherjee
Department of Neurosurgery, Post Graduate Institute of Medical Education and Research, Chandigarh, India
DOI:10.4103/2152-7806.172697
Copyright: © 2015 Surgical Neurology International This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.How to cite this article: Radotra BD, Salunke P, Parthan G, Dutta P, Vyas S, Mukherjee KK. True mycotic aneurysm in a patient with gonadotropinoma after trans-sphenoidal surgery. Surg Neurol Int 28-Dec-2015;6:193
How to cite this URL: Radotra BD, Salunke P, Parthan G, Dutta P, Vyas S, Mukherjee KK. True mycotic aneurysm in a patient with gonadotropinoma after trans-sphenoidal surgery. Surg Neurol Int 28-Dec-2015;6:193. Available from: http://surgicalneurologyint.com/surgicalint_articles/true-mycotic-aneurysm-in-a-patient-with-gonadotropinoma-after-trans%e2%80%91sphenoidal-surgery/
Abstract
Background:Immunosuppressive therapy, prolonged antibiotic use, and intrathecal injections are known risk factors for the development of invasive aspergillosis. Central nervous system (CNS) aspergillosis can manifest in many forms, including mycotic aneurysm formation. The majority of the mycotic aneurysms presents with subarachnoid hemorrhage after rupture and are associated with high mortality. Only 3 cases of true mycotic aneurysms have been reported following trans-sphenoidal surgery.
Case Description:A 38-year-old man was admitted with nonfunctioning pituitary adenoma for which he underwent trans-sphenoidal surgery. Three weeks later, he presented with cerebrospinal fluid (CSF) rhinorrhea and meningitis. He was treated with intrathecal and intravenous antibiotics, stress dose of glucocorticoids, and lumbar drain. The defect in the sphenoid bone was closed endoscopically. After 3 weeks of therapy, he suddenly became unresponsive, and computed tomography of the head showed subarachnoid hemorrhage. He succumbed to illness on the next day, and a limited autopsy of the brain was performed. The autopsy revealed extensive subarachnoid hemorrhage and aneurysmal dilatation, thrombosis of the basilar artery (BA), multiple hemorrhagic infarcts in the midbrain, and pons. Histopathology of the BA revealed the loss of internal elastic lamina and septate hyphae with an acute angle branching on Grocott's methenamine silver stain, conforming to the morphology of Aspergillus.
Conclusion:The possibility of intracranial fungal infection should be strongly considered in any patient receiving intrathecal antibiotics who fails to improve in 1–2 weeks, and frequent CSF culture for fungi should be performed to confirm the diagnosis. Since CSF culture has poor sensitivity in the diagnosis of fungal infections of CNS; empirical institution of antifungal therapy may be considered in this scenario.
Keywords: Aspergillosis, basilar artery, mycotic aneurysm, pituitary adenoma, subarachnoid hemorrhage, trans-sphenoidal surgery
INTRODUCTION
Aspergillosis of central nervous system (CNS) is an uncommon entity; however, the number of reported cases has increased in the past few years, possibly due to the widespread use of antibiotics, corticosteroids, and other immunosuppressants.[
CLINICAL PRESENTATION
A 38-year-old male presented with an episode of giddiness and uneasiness that lasted for 2 days. There was no history of dyspnea or palpitation. He did not have any comorbid illness. Clinical examination revealed bitemporal hemianopia and biochemical evaluation showed hyponatremia, hypocortisolism, and hypothyroidism, with elevated follicle-stimulating hormone, luteinizing hormone, and testosterone [
Three weeks later, he presented with intermittent fever and cerebrospinal fluid (CSF) rhinorrhea of 5 days duration. On examination, he had terminal neck rigidity. CSF studies were suggestive of pyogenic meningitis, CSF culture showed Enterobacter species, and he was started on appropriate antibiotics. The computed tomography (CT) scan showed pneumocephalus. Endoscopic repair of the defect was done after the infection was apparently controlled, and CSF was diverted through an external lumbar drain, to aid healing. Despite the repair, CSF rhinorrhea persisted, and CT scan showed the progression of pneumocephalus. Repeat CSF culture after a week grew Staphylococcus and he was started on intravenous Vancomycin and Meropenem with intrathecal Vancomycin. His preoperative total leukocyte count was 8700 mm3 with neutrophils 71%, lymphocytes 22%, monocytes 5%, and eosinophils 2%. At present admission, his total leukocyte count was 15,800 with neutrophils 82%, lymphocytes 14%, and monocytes 4%. His serum albumin and globulin were 4.5 g/dl and 3.5 g/dl, respectively. Serology for HIV was negative. The serial CSF reports of the patient are given in the
The cause of death was speculated to be due to rupture of an intracranial aneurysm. This may have been an association with pituitary macroadenoma (although it is seen often with functioning pituitary macroadenoma), or due to the weakening of vessel wall due to inflammatory exudates. The third possibility could be an inadvertent intraoperative injury such as snapping of a perforator from a distal vessel, resulting in pseudoaneurysm formation. To confirm the cause of death, a limited brain autopsy was performed, which revealed extensive subarachnoid hemorrhage and aneurysmal dilatation and thrombosis of the basilar artery (BA) [
Figure 4
Low power photo micrographs taken through the wall of the basilar artery at the site of aneurysm. (a) H and E staining showing dilated and destroyed arterial wall. (b) Elastic Van Gieson staining is showing the loss of internal elastic lamina on the upper part. (c) Gomorri methenamine silver staining is showing an invasive fungal elements from outer to the inner side in the aneurysmal wall. (d) High power photo micrograph showing numerous filamentary infiltrates with an acute angle branching confirming to the morphology of Aspergillus species
DISCUSSION
The common fungi associated with CNS infections include yeasts (Candida, Cryptococcus), moniliaceous molds (Aspergillus spp. and Fusarium spp.), dimorphic fungi (Blastomyces, Coccidioides, and Histoplasma), zygomycetes (Mucor spp., Rhizopus spp.), and dematiaceous fungi (Scedosporium spp.).[
Aspergillus is a ubiquitous fungus, and more than 180 species of Aspergillus have been described. Aspergillus fumigatus is the most common pathogen responsible for invasive Aspergillosis; although Aspergillus flavus, Aspergillus terreus, Aspergillus niger, and Aspergillus nidulans can also cause invasive disease.[
Mycotic aneurysms account for 0.7–5.4% of all intracranial aneurysms and is most commonly caused by bacteria; fungi are a rare cause of mycotic aneurysm.[
Postoperative mycotic aneurysms of the CNS are rare.[
Komatsu et al. have described the two processes of mycotic aneurysm formation; those secondary to meningitis and those secondary to sepsis. The former tend to affect the larger vessels and are a result of fungal invasion of vessels from outside, whereas the latter tend to involve peripheral vessels and are a result of the invasion of vessel wall from within.[
There are various possible portals through which the fungi would have entered into CNS. Hematogenous dissemination was unlikely as the histopathology showed that vessel was invaded from outside. The direct entry through the operative route was also unlikely, as the sphenoid mucosa showed no evidence of fungus; however, the possibility of subsequent lodgment and focal arachnoiditis due to the persistent CSF leak could not be excluded. Our patient received the intrathecal antibiotics, and this could have given the fungus access to CNS. Fungal meningitis has been reported after epidural steroids for pain.[
Aspergillus species may be found as a commensal in paranasal sinuses.[
High index of the suspicion based on clinical, radiological, and fungal culture is essential to make a diagnosis of CNS aspergillosis, and brain biopsy is required for the definitive diagnosis, which is not feasible in most of the cases.[
CONCLUSION
The possibility of intracranial fungal infection should be strongly considered in any patient receiving intrathecal antibiotics who fails to improve in 1–2 weeks, and frequent CSF culture for fungi should be performed to confirm the diagnosis. Since CSF culture has poor sensitivity in the diagnosis of the fungal infections of CNS, the empirical institution of antifungal therapy may be considered in this scenario.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
References
1. Ducruet AF, Hickman ZL, Zacharia BE, Narula R, Grobelny BT, Gorski J. Intracranial infectious aneurysms: A comprehensive review. Neurosurg Rev. 2010. 33: 37-46
2. Horten BC, Abbott GF, Porro RS. Fungal aneurysms of intracranial vessels. Arch Neurol. 1976. 33: 577-9
3. Hot A, Mazighi M, Lecuit M, Poirée S, Viard JP, Loulergue P. Fungal internal carotid artery aneurysms: Successful embolization of an Aspergillus-associated case and review. Clin Infect Dis. 2007. 45: e156-61
4. Hurst RW, Judkins A, Bolger W, Chu A, Loevner LA. Mycotic aneurysm and cerebral infarction resulting from fungal sinusitis: Imaging and pathologic correlation. AJNR Am J Neuroradiol. 2001. 22: 858-63
5. Iihara K, Makita Y, Nabeshima S, Tei T, Keyaki A, Nioka H. Aspergillosis of the central nervous system causing subarachnoid hemorrhage from mycotic aneurysm of the basilar artery – Case report. Neurol Med Chir (Tokyo). 1990. 30: 618-23
6. Kasper D, Fauci A, Hauser S, Longo D, Jameson J, Loscalzo J.editors. Diagnosis and treatment of fungal infections. Harrison's Principles of Internal Medicine. New York: McGraw-Hill; 2015. I: 1239-
7. Kleinfeld K, Jones P, Riebau D, Beck A, Paueksakon P, Abel T. Vascular complications of fungal meningitis attributed to injections of contaminated methylprednisolone acetate. JAMA Neurol. 2013. 70: 1173-6
8. Kleinschmidt-DeMasters BK. Central nervous system aspergillosis: A 20-year retrospective series. Hum Pathol. 2002. 33: 116-24
9. Komatsu Y, Narushima K, Kobayashi E, Tomono Y, Nose T. Aspergillus mycotic aneurysm – Case report. Neurol Med Chir (Tokyo). 1991. 31: 346-50
10. Kousha M, Tadi R, Soubani AO. Pulmonary aspergillosis: A clinical review. Eur Respir Rev. 2011. 20: 156-74
11. Masago A, Fukuoka H, Yoshida T, Majima K, Tada T, Nagai H. Intracranial mycotic aneurysm caused by Aspergillus – Case report. Neurol Med Chir (Tokyo). 1992. 32: 904-7
12. McCarthy M, Rosengart A, Schuetz AN, Kontoyiannis DP, Walsh TJ. Mold infections of the central nervous system. N Engl J Med. 2014. 371: 150-60
13. Mielke B, Weir B, Oldring D, von Westarp C. Fungal aneurysm: Case report and review of the literature. Neurosurgery. 1981. 9: 578-82
14. Mukoyama M, Gimple K, Poser CM. Aspergillosis of the central nervous system. Report of a brain abscess due to A. fumigatus and review of the literature. Neurology. 1969. 19: 967-74
15. Murthy JM. Fungal infections of the central nervous system: The clinical syndromes. Neurol India. 2007. 55: 221-5
16. Okada Y, Shima T, Nishida M, Yamane K, Yoshida A. Subarachnoid hemorrhage caused by Aspergillus aneurysm as a complication of transcranial biopsy of an orbital apex lesion – Case report. Neurol Med Chir (Tokyo). 1998. 38: 432-7
17. Redmond A, Dancer C, Woods ML. Fungal infections of the central nervous system: A review of fungal pathogens and treatment. Neurol India. 2007. 55: 251-9
18. Ruhnke M, Kofla G, Otto K, Schwartz S. CNS aspergillosis: Recognition, diagnosis and management. CNS Drugs. 2007. 21: 659-76
19. Segal BH, Walsh TJ. Current approaches to diagnosis and treatment of invasive aspergillosis. Am J Respir Crit Care Med. 2006. 173: 707-17
20. Sugui JA, Kwon-Chung KJ, Juvvadi PR, Latgé JP, Steinbach WJ. Aspergillus fumigatus and related species. Cold Spring Harb Perspect Med. 2014. 5: a019786-
21. Vennewald I, Henker M, Klemm E, Seebacher C. Fungal colonization of the paranasal sinuses. Mycoses. 1999. 42 Suppl 2: 33-6
22. Visudhiphan P, Bunyaratavej S, Khantanaphar S. Cerebral aspergillosis.Report of three cases. J Neurosurg. 1973. 38: 472-6
23. Walsh TJ, Mendelsohn G. Invasive aspergillosis complicating Cushing's syndrome. Arch Intern Med. 1981. 141: 1227-8