- Department of Neurosurgery, National Institute of Neurology and Neurosurgery, Mexico City, Mexico.
- Department of Neuroinfectology, National Institute of Neurology and Neurosurgery, Mexico City, Mexico.
Marcos V. Sangrador-Deitos, Department of Neurosurgery, National Institute of Neurology and Neurosurgery, Mexico City, Mexico.
DOI:10.25259/SNI_469_2023Copyright: © 2023 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, transform, 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: Jorge Armando Lara-Olivas1, Marcos V. Sangrador-Deitos1, Ricardo Marian-Magaña1, Karen Elizabeth Padilla-Leal1, José L. Soto-Hernández2, Juan L. Gomez-Amador1. Endoscopic endonasal resection of a Drechslera hawaiiensis sphenoid fungal ball. 08-Sep-2023;14:321
How to cite this URL: Jorge Armando Lara-Olivas1, Marcos V. Sangrador-Deitos1, Ricardo Marian-Magaña1, Karen Elizabeth Padilla-Leal1, José L. Soto-Hernández2, Juan L. Gomez-Amador1. Endoscopic endonasal resection of a Drechslera hawaiiensis sphenoid fungal ball. 08-Sep-2023;14:321. Available from: https://surgicalneurologyint.com/surgicalint-articles/endoscopic-endonasal-resection-of-a-drechslera-hawaiiensis-sphenoid-fungal-ball/
Background: Fungal infections should always be considered in difficult-to-treat paranasal sinus conditions. Sphenoid fungal balls are characterized by the presence of dense fungal masses in the sinus cavity without invasion of surrounding tissues. This case emphasizes the importance of accurate terminology and management and also highlights the involvement of rare pathogens such as Drechslera hawaiiensis. Diagnosis is typically based on imaging studies and intraoperative findings. Accurate identification of the pathogen is crucial. Fungal infections of the paranasal sinuses, including fungus balls, can present challenges in diagnosis and treatment. D. hawaiiensis, although infrequent, can cause potential life-threatening infections.
Case Description: We present a 26-year-old non-HIV male patient who presented with nasal symptoms and mild headaches. The patient underwent an endoscopic exploration that revealed a soft, grayish lesion with a buttery consistency. Gross total resection was achieved and the lesion was identified as being caused by D. hawaiiensis; thus, intravenous antifungal treatment was given.
Conclusion: Endoscopic surgery remains the preferred approach for disease control. Considering alternative treatments and exploring novel approaches are essential in managing complex pathologies in neurosurgical practice.
Keywords: Bipolaris, Drechslera hawaiiensis, Endoscopic endonasal resection, Mycotic, Skull base surgery
Sphenoid fungus ball refers to the collection of dense fungal masses in the sinus cavity with no invasion of surrounding tissues. The clinical presentation is non-specific and the diagnosis is based on imaging studies. Surgical treatment, often performed using an endoscopic endonasal approach, is usually curative. Fungi may play a significant role in chronic sinusitis and central nervous system infections, as they can be cultured using sensitive methods in over 95% of patients.[
It is important to avoid using misleading terms such as aspergilloma, aspergillosis, or mycetoma as there are different pathogens involved, such as D. hawaiiensis in this particular case. The latter is a rapidly-growing fungus that produces a grayish colony; the hyphae, conidiophores, and conidia are dematiaceous and are found worldwide in the air, florae, grasslands, grains, rotten food, and soil. When affecting humans, this pathogen can produce subcutaneous or systemic infectious disease.[
Drechslera sp. is commonly found in the soil of tropical climates and hardly causes human infection. When this occurs, it usually affects immunocompromised individuals. These fungi are usually soil saprophytes and plant parasites, possessing a low pathogenic capacity; nevertheless, they are capable of invading the tissues of individuals with impaired resistance when they inhale particles from sources such as vegetables, soil, plants, insect bites, wounds, and minor trauma, causing infection to the host.[
We present a 26-year-old male patient, from a rural location in Baja California, Mexico, with no relevant background, presenting with mild intermittent headaches, nasal congestion, rhinorrhea, labored breathing, and anosmia. However, no cacosmia, visual disturbances, facial deformities, or meningeal signs were present. He was previously misdiagnosed with an esthesioneuroblastoma based on imaging studies. Magnetic resonance imaging (MRI) revealed an isodense lesion that appeared irregular and homogeneous, with peripheral enhancement on T1 weighted image with contrast. Invasion of the ethmoid and sphenoid sinuses with a necrotic center was observed. On T2WI, a highly hypointense appearance of the lesion suggests an infectious etiology [
Preoperative T1WI with contrast magnetic resonance imaging: (a) axial, (b) coronal, and (c) sagittal planes in which an isodense irregular lesion can be seen completely invading the sphenoid and ethmoid sinuses. Gadolinium enhancement shows ring enhancement. (d) T2WI sequence revealed a highly hypointense lesion, which is suggestive of an infectious etiology.
Using a 0° angled rigid endoscope, the right nostril was approached first. The lesion was immediately reached, as it was coming out of the middle meatus which was completely occupied by the tumor. The soft and grayish lesion with a very buttery consistency was easily resected using biopsy rongeurs and ringed curettes. As resection continued, this buttery consistency was notorious and this deemed the resection quite simple. Once the middle meatus was deemed completely clean, a right turbinectomy was performed to access the sphenoid and maxillary sinuses. As the lesion had completely destroyed the rostrum sphenoidale, no proper bony landmarks, such as the ostium sphenoidale, were found. Resection of all the reachable portions located within the sphenoid sinus was accomplished through the right nostril first. Subsequently, the left nostril was approached and a left turbinectomy was performed as well. As mentioned, the anterior wall of the sphenoid sinus was completely destroyed as well as the posterior portion of the nasal septum; thus, no posterior septectomy was needed, and access to the sphenoid sinus was quite straightforward. Once gross total resection of the lesion was achieved, bony landmarks within the sphenoid sinus were observed; being the sella turcica and clival recess limited laterally by the parasellar and paraclival segments of the internal carotid artery (ICA), respectively. Neuronavigation was used to confirm the lateral and inferior limits of the lesion located in the clival recess. After resection of the sphenoidal portion of the lesion, bilateral maxillostomies were performed to access the maxillary sinuses. Finally, hemostasis was performed by the placement of gel foam on the surgical site until no active sites of bleeding were observed [
The resected material was sent to the microbiology laboratory for fungal studies. Smear preparations with lactophenol cotton blue stain revealed hyphae elements. Cultures on Sabouraud’s dextrose agar presented the proliferation of a dematiaceous fungus displaying numerous multi-septate spores. The fungus was determined to be D. hawaiiensis, due to its morphological appearance[
(a) Micromorphology of the filamentous fungus Drechslera hawaiiensis using lactophenol cotton blue stain. Hyaline fungi can acquire the blue hue from lactophenol, whereas dematiaceous fungi will exhibit a brown discoloration caused by the mold’s pigment production. (b) Macroscopic sample of the thick, greasy, and dark material extracted from the antrum. There was an abundance of dark brown mucous which possessed a smooth and buttery texture, along with buttery consistency and grayish color.
The utilization of nasal endoscopy and computed tomography scans has increased the frequency of diagnosis for fungal diseases of the paranasal sinuses, including fungus balls.[
A fungus ball is a complex condition that can be categorized into invasive and non-invasive types.[
Caution should be exercised when immunocompetent individuals exhibit symptoms suggestive of fungal diseases, with risk factors including exposure to the fungus, particularly in endemic areas. Numerous conditions such as esthesioneuroblastoma, sinusitis, chordoma, tension headache, vascular headache, foreign bodies, brain abscess, epidural abscess, meningitis, and subdural abscess should be considered as part of the differential diagnosis based on imaging studies.[
The patient’s prior misdiagnosis several months before the consultation proved to be a significant distractor, leading to a chain reaction of colleagues presuming that esthesioneuroblastoma or a neoplasm was the sole possibility. This presumption could have delayed an earlier intervention and developed an unfavorable outcome. The patient’s rural background suggests that fungal soil may be prevalent in that region. Since the complete surgical removal of infected tissue may not be feasible, topical corticosteroid aerosols should be used under antimycotic coverage to prevent recurrence.[
The patient did not experience any sequelae following the surgery. To consider unusual and infrequent alternative diagnoses, such as invasive fungal pathology with an uncommon germ is necessary to achieve a successful medical diagnosis. Therefore, it is necessary to implement diligent monitoring, including the potential for recurrent surgery to extract infected tissues, as well as providing comprehensive postoperative care at the local level. It is critical to always consider a rare pathogen in any sinonasal pathology, particularly when it concerns the anterior skull base. Failure to make a prompt diagnosis may result in a poor outcome, while, when an adequate diagnosis and medical management are performed, good outcomes are usually achieved [
Fungal pathogens have the potential to imitate neoplastic processes, and detecting them may require multiple analyses as they may not be easily isolated in a single test or detected through histology. D. hawaiiensis is an infrequent pathogen but potentially life-threatening when not identified correctly. Fungus balls can result in secondary mycotic manifestations, which can be fatal if not treated effectively. Management of these infections usually requires local surgical excision and prolonged courses of antifungal agents to prevent recurrence.
Patient’s consent not required as patient’s identity is not disclosed or compromised.
There are no conflicts of interest.
The author(s) confirms that there was no use of artificial intelligence (AI)-assisted technology for assisting in the writing or editing of the manuscript and no images were manipulated using AI.
The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.
We, the authors, appreciate the valuable contribution of Dr. Edith Sánchez Paredes from the UNAM School of Medicine Microbiology Department, who was responsible for the microbiological identification of the fungus, and kindly provided the microphotograph depicting this microorganism.
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