Journal of Otology & RhinologyISSN: 2324-8785

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Case Report, J Otol Rhinol Vol: 5 Issue: 4

Invasive Fugal Sinusitis Causing Massive Epistaxis: A Case Report

Rovira A*, Suarez G, Gonzalez-Compta and Cisa E
Depratment of Otorhinolaryngology, Hospital Universitari de Bellvitge, L Hospitalet de Llobregat, Barcelona, Spain
Corresponding author : Rovira A
Department of Otorhinolaryngology, Hospital Universitari de Bellvitge, L Hospitalet de Llobregat, Barcelona, Spain, C/feixa lalrga s/n. 09807
Tel: +34678960441
E-mail: [email protected]
Received: May 02, 2016 Accepted: May 31, 2016 Published: June 05, 2016
Citation: Rovira A, Suarez G, Gonzalez-Compta, Cisa E (2016) Invasive Fungal Sinusitis Causing Massive Epistaxis: A Case Report. J Otol Rhinol 5:4. doi:10.4172/2324-8785.1000283

Abstract

Background: Invasive fungal sinusitis (IFS) is a rare entity. An increase of IFS cases is observed as the number of immunosuppressive conditions are rising. Clinical suspicion is paramount due to the rapid and aggressive behaviour of this condition. Early surgical debridement and antifungal therapy correspond the first treatment option. Overall survival rate has been reported between 50-100%. Internal carotid artery rupture is a rare complication that may cause death; when present, the endovascular approach seems to be the best option for treatment.

Case presentation: A 64-year-old man with chronic obstructive pulmonary disease presented at emergency room complaining headache and diplopia. He has been treated with intranasal and systemic corticosteroids five months ago due to an acute exacerbation of his pulmonary disease. CT-scan showed sphenoid sinus opacification with erosion of the lateral wall, superior and inferior orbital fissure. Histopathology analysis of sinus mucosa identified microorganisms morphologically compatible with mucor. After one week of treatment with liposomal Amphotericin B the patient presented to the emergency room (ER) with massive epistaxis. Anterior and posterior nasal packing was performed and the imaging revealed a carotid-cavernous fistula. A left internal carotid artery occlusion was performed, with a consequent left hemi cerebral infraction.

Conclusions: IFS may rapidly evolve to fatal consequences. Clinical suspicion in immunosuppressed patients is of critical importance as rapid and aggressive treatment may be lifesaving when facing IFS.
 

Keywords: Sphenoid mucormicosis; Internal carotid artery rupture; Epistaxis

Keywords

Sphenoid mucormicosis; Internal carotid artery rupture; Epistaxis

Background

Invasive fungal sinusitis (IFS) is a rare entity. An increase of IFS cases is observed as the number immunosuppressive conditions are rising. IFS is a potentially fatal disease, especially when sphenoid sinus is involved due to the important surrounding structures. Patients with sphenoid lesions at early stage are usually asymptomatic or suffer unspecific symptoms, thereby, delaying the diagnosis. Given the IFS high mortality, early recognition and treatment are essential for increasing the patient’s survival. In the early stage the most common symptoms are diffuse headache and rhinorrhea. Visual disturbance, retro‐orbital and frontal headache, ophthalmoplegia, ptosis, facial and orbital pain may appear as the disease advance [1,2].
In the immunosuppressed patient a high clinical suspicion should proceed with a full otorhinolaryngological exploration including nasal endoscopic examination. Tissue biopsy and cultures are mandatory. Computed tomography (CT) should be the first imaging test. Sinus opacification, calcification or bony destruction although nonspecific may be present when the sphenoid sinus is involved, special attention to the superior and lateral walls in order to rule out intracranial extension. In a recent study with twelve patients enrolled, 8 suffered intracranial invasion, and MRI was superior to CT in detecting extrasphenoidal lesions [1].
Although the potential risk of injury to the internal carotid artery, cavernous sinus or optic nerve, early surgical debridement is of utmost importance in the IFS treatment. Antifungal therapy must be administrated, being Liposomal Amphotericin B the first option at a dose between 3-5 mg/kg/d until an accumulative dose of 2g is achieved [3,4].
Overall survival rate has been reported in a range between 50‐100% [1,57].
Internal carotid artery (ICA) rupture is a rare complication of IFS. When present, the best treatment option is an endovascular approach with occlusion of the affected segment followed by aggressive debridement and antifungal therapy.

Case Presentation

A 64‐year‐old man with a four month history of headaches and recent onset of diplopia was admitted to our hospital. The patient’s medical history was relevant for arterial hypertension and chronic obstructive pulmonary disease. He suffered an acute exacerbation of his pulmonary disease five months ago and was treated with antibiotics, short‐acting bronchodilators and oral corticosteroids. CT‐scan showed a left sphenoid and ethmoidal sinus opacification with bony erosion of the lateral wall of the sphenoid sinus and superior and inferior orbital fissure (Figure 1). The differential diagnosis included tumor, bacterial sinusitis and invasive fungal sinusitis. Neurologic examination revealed decreased left extraocular motion and eyelid ptosis and binocular diplopia due to cranial nerves III and VI paresis. The visual acuity was found normal.
Figure 1: CT-scan showing left sphenoid and ethmoidal sinus opacification.
Endoscopic endonasal examination did not reveal any abnormality. Laboratory evaluation showed a white blood cell count of 11.300/L, hemoglobin13.1 g/dL, platelet count of 334.000/L and negative serology for HBC, HCV, HIV 1+2, LUES. The patient was taken to the operating room for an endoscopic exploration under general anaesthesia. A left sphenoid sinus mucosa swelling was identified being more predominant towards the lateral wall without any other pathologic findings. Mucosal samples were sent for microbiologic and pathologic analysis. Aerobic, anaerobic and fungal cultures were negative as well as aspergillus, pan fungal and tuberculosis PCR. Pathological analysis showed chronic inflammation with necrotic material and microorganisms morphologically compatible with mucor. After the sphenoidotomy and medical treatment with liposomal Amphoterincin B 5mg/kg/d, the patient presented clinical improvement with headache resolution and diplopia improvement. Given the good clinical evolution, the patient was discharged with home IV Amphotericin treatment. The patient returned to the emergency room one week after being discharged with massive nasal bleeding, requiring anterior and posterior bilateral nasal packing. A left ICA rupture was suspected and patient was transferred to interventional neuroradiology theatre were active bleeding from the aforementioned artery was identified through a carotid‐cavernous fistula. The left ICA was embolized with complete occlusion and bleeding cessation. Posterior CT scans showed complete left cerebral infraction and life support was withdrawn leading to a fatal end after one day.

Conclusions

IFS may rapidly evolve to fatal consequences. Clinical suspicion in immunosuppressed patients is of critical importance as rapid and aggressive treatment may be lifesaving when facing IFS.

Authors Contributions

AR and GS attended the patient in the emergency room and have contributed equally in writing the manuscript. XG and EC performed the sample collection and the follow-up of the patient. All authors read and approved the final manuscript.

Acknowledgments

We would like to thank the staff of the Bellvitge University Hospital and the Patient’s relatives who were kind enough to give their consent for this case report.

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