- Department of Biomedicine, Neurosciences and Advanced Diagnostic, Section of Neurosurgery, University of Palermo, Palermo, Italy.
- Department of Health Promotion Sciences, Section of Infectious Diseases, University of Palermo, Palermo, Italy.
- Department of Health Promotion Sciences, Pathology Unit, University of Palermo, Palermo, Italy.
- Department of Diagnostic and Therapeutic Services, and Neurology Service, Mediterranean Institute for Transplantation and Advanced Specialized Therapies, Radiology Service, University of Palermo & ISMETT, Palermo, Italy.
Department of Biomedicine, Neurosciences and Advanced Diagnostic, Section of Neurosurgery, University of Palermo, Palermo, Italy.
DOI:10.25259/SNI_805_2020Copyright: © 2021 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 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: Laura Brunasso1, Roberta Costanzo1, Antonio Cascio2, Ada Florena3, Gianvincenzo Sparacia4, Domenico Gerardo Iacopino1, Giovanni Grasso1. Seizure in isolated brain cryptococcoma: Case report and review of the literature. 14-Apr-2021;12:153
How to cite this URL: Laura Brunasso1, Roberta Costanzo1, Antonio Cascio2, Ada Florena3, Gianvincenzo Sparacia4, Domenico Gerardo Iacopino1, Giovanni Grasso1. Seizure in isolated brain cryptococcoma: Case report and review of the literature. 14-Apr-2021;12:153. Available from: https://surgicalneurologyint.com/?post_type=surgicalint_articles&p=10720
Background: Central nervous system (CNS) cryptococcosis is an invasive fungal infection predominantly seen among immunosuppressed patients causing meningitis or meningoencephalitis. Rarely, cryptococcosis can affect immunologically competent hosts with the formation of localized CNS granulomatous reaction, known as cryptococcoma. Common symptoms of CNS cryptococcoma are headaches, consciousness or mental changes, focal deficits, and cranial nerve dysfunction. Rarely, seizures are the only presenting symptom.
Case Description: We report the case of an immunocompetent patient with a solitary CNS cryptococcoma presenting with a long history of non-responsive generalized seizure who has been successfully operated.
Conclusion: CNS cryptococcoma is a rare entity, and in immunocompetent patients, its diagnosis can be challenging. The pathophysiology of lesion-related seizure is discussed along with a review of the pertinent literature.
Keywords: Central nervous system, Cryptococcoma, Seizure
Cryptococcosis is an invasive fungal infection caused by two species of Cryptococcus spp. Cryptococcus neoformans is commonly related to immunocompromised patients mainly causing meningitis. Cryptococcus gattii is encountered in immunocompetent population and it is associated with cryptococcoma formation in the brain and lungs.[
Here, we present the case of an intracranial cryptococcoma in a 32-year-old woman with normal immunity. The main mechanisms underlying seizure manifestation along with a review of the pertinent literature are reported.
A 32-year-old Indian woman presented with a history of tonic-clonic seizures since 2013, started during her first pregnancy. In 2016, she underwent electroencephalogram which confirmed the diagnosis of epilepsy. Accordingly, she started antiepileptic treatment with levetiracetam at initial daily dose of 1 g. Due to the recurrence of seizures, the drug was increased till a daily dose of 1.5 g with a poor seizure control. In 2019, a brain magnetic resonance (MR) examination revealed a right temporo-mesial lesion with an irregular peripheral contrast enhancement. The lesion appeared to protrude toward the right cerebral peduncle with brainstem compression, highly suggestive of low-grade glioma. MR spectroscopy supported the suspicion of glioma. The patient underwent functional MR showing anterior dislocation of the inferior longitudinal fasciculus. [
Pre-operative magnetic resonance (MR) spectroscopy (a) depicts a low N-acetylaspartate (NAA) with choline (Cho)/NAA of 1.33 compared to normal brain parenchyma, suggestive for low-grade glioma; functional MR (b) showing the anterior dislocation of the inferior longitudinal fasciculus (ILF) (red tract) and a unaffected corticospinal tracts (blue tracts).
The procedure was performed by the use of neuronavigation. A right temporal craniotomy was performed. Through a transulcal approach, the lesion was reached. The lesion appeared as a calcified mass tenaciously attached to the contiguous structures. The lesion was entered, and a yellow-like material was found densely packing the mass. After a careful debulking, the capsule was removed in fragments except for its medial part being strictly adherent to the brainstem [
Histopathology showed multiple yeasts consistent with Cryptococcus spp. strongly embedded into an amorphous eosinophilic fibrillar material. Period Acid–Schiff and mucicarmine stain revealed purple organisms and numerous budding yeasts consistent with Cryptococcus spp. [
After surgery, the patient presented with a mild left leg coordination impairment which disappeared in a few days. Forty-four hours post-operative MRI showed a residual capsule fragment adherent to the midbrain [
Cryptococcal infection occurs by inhalation of infectious propagules from environmental reservoirs and pulmonary impairment constitutes the first manifestation.[
It has been reported that Cryptococcus spp. enters the Virchow–Robin spaces and gives rise to small cysts in the brain parenchyma, inducing a chronic granulomatous reaction composed by macrophages, lymphocytes, and foreign body-type giant cells.[
Solitary cryptococcoma is a rare lesion,[
In CNS cryptococcal infection, a combined medical and surgical approach is considered the optimal treatment.[
According to our experience and based on the few reports available, we speculate that surgical resection should be always attempted to reach a firm diagnosis and brain decompression.[
CNS cryptococcoma is a rare entity and could affect immunocompetent individuals. It has no specific radiologic findings and can mimic CNS tumors. Our experience suggests that in immunocompetent patients, an isolated intracerebral cryptococcal granuloma can be challenging to diagnose where seizure is the only presenting symptom. In these cases, surgical treatment is mandatory for the diagnosis and seizure resolution.
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