Abstract
Isolated disease of sphenoid sinus is rare, representing 2-3% of all paranasal sinus lesions. Usually it is inflammatory in origin; very rarely it is due to neoplasm. Isolated sphenoid sinus diseases are difficult to diagnose and to treat because either the symptoms are very vague or they present to us very late as a result of disease complications. Here we are presenting a case of isolated sphenoid fungal sinusitis. A 40 year female came to our ENT outpatient department with complaints of intermittent headache for past 3 months. She was treated for similar complaints by a general practitioner with antibiotics and analgesics before three months. She was referred to an ophthalmologist and neurologist as the headache did not subside. Since there was no abnormality in ophthalmological examination, the neurologist suggested a MRI which showed opacification of the sphenoid sinus. Hence she was referred to an ENT specialist. ENT Clinical examination was normal. Urgent CT scan along with MRI was ordered which revealed complete opacification of the sphenoid sinus. There was no air fluid level, no hyper dense foci suggestive of fungal elements. There was no bony erosion. A provisional diagnosis of Chronic Sphenoid sinusitis was made and planned for endoscopic sphenoidotomy under general anesthesia. Sphenoid sinus was opened and fungal debri was seen inside, which was sent for culture. Sphenoid ostium was widened. Post operatively patient was completely relieved of headache. Patient was started on Tab.Itraconazole 200mg/day for 6 weeks, since the culture was suggestive of aspergillosis. Owing to the non specific presentation and the vague symptomology of the disease there may be considerable delay in diagnosing and treating the patient. Hence imaging studies like CT and MRI is necessary when the disease is suspected for prompt diagnosis.
Keywords: Isolated sphenoid sinusitis, Fungal sinusitis, Endoscopic sinus surgery, Imaging studies
Introduction
Isolated disease of sphenoid sinus is rare, representing 2–3% of all paranasal sinus lesions [1, 2]. Usually it is inflammatory in origin, very rarely it is due to neoplasms [3, 4]. Isolated sphenoid sinus diseases are difficult to diagnose and to treat because either the symptoms are very vague or they present to us very late as a result of disease complications. Nevertheless prompt diagnosis and early intervention is mandatory as any delay can lead onto catastrophic consequences because of contiguity to vital structures like optic nerve, internal carotid artery, cavernous sinus, cranial nerves. Here we are presenting a case of isolated sphenoidal fungal sinusitis.
Case Report
A 40 year female came to our ENT outpatient department with complaints of intermittent headache for past 3 months. Headache was intermittent, more in the vertex, not associated with vomiting, visual symptoms. There was history of rhinorrhoea, nasal obstruction and nasal discharge.
Past history revealed that she was treated for similar complaints by a general practitioner with antibiotics and analgesics before 3 months. She was referred to an ophthalmologist and neurologist as the headache did not subside. Since there was no abnormality in ophthalmological examination, the neurologist suggested an MRI which showed opacification of the sphenoid sinus. Hence the patient was suggested to visit an otorhinolaryngologist but she was not willing as the patient had temporary pain relief with analgesics.
Since the patient again developed continuous intolerable headache for past 2 weeks which was not relieved by usual analgesics, she presented to us in our ENT-OPD. Clinical examination with anterior and posterior rhinoscopy was normal. Neurological examination was normal. Diagnostic nasal endoscopy revealed no abnormalities. Urgent CT scan was ordered which revealed complete opacification of the sphenoid sinus. There was no air fluid level, no hyperdense foci suggestive of fungal elements. MRI Brain was also taken to confirm the CT findings and to make sure if any other soft tissue involvement was overlooked. MRI was also in concordance with the CT findings showing no air fluid level, no hyperdense foci and no bony erosion as seen in Fig. 1.
Fig. 1.

MRI image of brain at the level of sphenoid sinus with arrow mark showing bony opacification of sphenoid sinus
Figures 2 and 3 showing the CT image of the sphenoid bone—axial and coronal view respectively without any air fluid level or hyperdense foci.
Fig. 2.

CT axial view
Fig. 3.

CT coronal view
A provisional diagnosis of chronic sphenoidal sinusitis was made and planned for endoscopic sphenoidotomy under general anesthesia which was considered the optimal treatment [5]. Endoscopic sphenoidotomy was done through natural ostium of sphenoid. Mucopus was drained, then to our surprise, there was fungal debri seen inside the sphenoid sinus, which was sent for culture. Sphenoid ostium was widened as shown in Fig. 4. Post operatively patient was completely relieved of headache.
Fig. 4.

Endoscopic picture of the sphenoid sinus with arrow mark showing the fungal debri within the sphenoid sinus with the sphenoid sinus ostium widened
Patient was started on Tab.Itraconazole 200 mg/day for 6 weeks, since the culture was suggestive of aspergillosis [6]. Post operatively patient was completely relieved of pain and sphenoid ostium was patent.
Discussion
Isolated sphenoid sinus disease which is rare can be classified into inflammatory and non-inflammatory lesions. Inflammatory lesions include bacterial, fungal sphenoiditis, mucocele, sphenochoanal polyp. Non inflammatory lesions include benign neoplasms like inverted papilloma, fibrous dysplasia, squamous cell carcinoma of sphenoid sinus, CSF leak, and internal carotid artery aneurysms.
The initial symptoms of isolated sphenoid sinus disease are vague, making it difficult to diagnose. Most common symptom—usually headache of various intensity and different locations [7, 8], usually limited to the vertex. Since a patient with headache is usually treated by a general practitioner and other non ENT personnel like neurologist, neurosurgeon and ophthalmologist there can be significant delay in arriving at a correct diagnosis and prompt intervention.
But Sieskiewicz et al. [9] showed that there was no significant delay between otorhinolaryngologist (ORL) group and non otorhinolaryngologist (non-ORL) group as the mean duration of symptoms in non-ORL group is 9.5 months and ORL group is 10.8 months.
Sieskiewicz et al. also showed that the mean time for the first medical assessment to imagery studies is less in non-ORL group (1.8 months) as compared to ORL group (4.1 months). This maybe due to otorhinolaryngologists as they can be misled by a normal conventional X ray of PNS and normal endoscopic findings.
Endoscopic examination of the sphenoethmoidal recess can be normal in chronic inflammatory lesions of sphenoid making the diagnosis difficult. Hence diagnosis of sphenoid sinus lesions are based mainly on imaging studies.
Complications
Iatrogenic damage to the cavernous part of carotid artery and optic nerve while removing the sphenoid intersinus septum. Excess enlargement of the sphenoidotomy inferiorly with sphenoid punch leads to damage of sphenopalatine artery.
Conclusion
Isolated sphenoidal disease though rare is diagnosed much frequently now days. Owing to the non specific presentation and the vague symptomology of the disease there may be considerable delay in diagnosing and treating the patient. Hence imaging studies like CT and MRI is necessary when the disease is suspected for prompt diagnosis and once diagnosed if surgery is indicated it should be done as it the best optimal treatment.
Compliance with Ethical Standards
Conflict of interest
Dr. RajPrakash Dharmapuri Yaadhava Krishnan, Dr. Pragadeeswaran Kumarasekaran and Dr. Roopak Visakan Raja declare that they have no conflict of interest.
Contributor Information
RajPrakash Dharmapuri Yaadhava Krishnan, Email: dyrajprakash@gmail.com.
Pragadeeswaran Kumarasekaran, Email: prage.k@gmail.com.
Roopak Visakan Raja, Email: fantazrup11@gmail.com.
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