Aspergillosis: An Unusual Case of Orbital Apex Syndrome

Kaeser, Pierre-François; Borruat, F.–X.

doi:10.1055/s-0029-1245214

Cited by 2 publications

(1 citation statement)

References 4 publications

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“…Unfortunately, permanent blindness [ 9 , 16 – 18 ] and high mortality rates [ 19 – 22 ] are reported even after immediate antifungal therapy. Amphotericin B is most used in sino-orbital aspergillosis, 40% to 60% of patients being responsive to it.…”

Section: Discussionmentioning

confidence: 99%

Orbital apex syndrome secondary to aspergilloma masquerading as a paranasal sinus tumor

Chang¹,

Chang²,

Chien³

et al. 2018

Medicine

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Rationale:Orbital apex syndrome is a complex clinical disorder featuring a collection of cranial nerve deficits characterized by impairment of the extraocular muscles, the ophthalmic branch of the trigeminal nerve, and even the optic nerve. Sino-orbital aspergillosis is rare but aggressive infection. Surgical resection accompanied by antifungal medication is advised currently.Patient concerns:We report a 61-year-old woman diagnosed as aspergilloma presenting with the characteristic manifestations and imaging features of orbital apex syndrome.Diagnoses:Paranasal sinus tumor was misdiagnosed initially according to magnetic resonance imaging of the orbit. Finally aspergilloma was diagnosed by pathologic report.Interventions:The anti-fungal medication, voriconazole, was administered immediately. Surgical excision was also done due to the poor response to medical treatment.Outcomes:Postoperative follow-up showed no recurrence of aspergillosis but the vision was lost permanently.Lessons:Invasive sino-orbital aspergillosis as an aggressive disease with highly invasive patterns and it may be misdiagnosed as tumors. To achieve better prognosis and survival, clinicians should be aware of this distinct manifestation.

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Section: Discussionmentioning

confidence: 99%

Orbital apex syndrome secondary to aspergilloma masquerading as a paranasal sinus tumor

Chang¹,

Chang²,

Chien³

et al. 2018

Medicine

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Orbital Apex Syndrome Secondary to Invasive Aspergillus Infection: A Case Series and Literature Review

Yuan¹,

Tandon²,

Li³

et al. 2020

Journal of Neuro-Ophthalmology

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Background: Invasive fungal sinusitis carries high morbidity and mortality and often poses a diagnostic challenge. Orbital apex syndrome (OAS) is not an uncommon presentation in the setting of invasive fungal sinusitis. Delays in diagnosis and appropriate treatment can result in permanent visual dysfunction and, potentially, death. We present 2 cases of OAS secondary to invasive sinus aspergillosis, detailing the diagnostic process, treatment, and outcome for both patients. Subsequently, we present a review of the literature and combined analysis of our 2 patients plus 71 cases from previously published reports. Methods: Literature review was performed to identify demographic, diagnostic, clinical, and treatment data of patients with OAS caused by Aspergillus species. Results: The review resulted in 52 included articles with 71 patients, plus our 2 reported patients, leading to a total of 73 subjects included in the analysis. The average age of patients at presentation was 59.9 years. A combination of visual disturbance and pain (headache and/or periocular pain) was the most common presentation reported (46 cases; 63%). Diabetes mellitus was reported in 15 cases (21%), with more than half specifically noted to have poorly controlled diabetes. After diabetes, the second most common cause of immunocompromise was chronic steroid use (n = 13; 18%). Empiric antifungal treatment was started in 10 patients (14%), while 25 patients (34%) were first treated with systemic steroids due to a concern for an inflammatory etiology. Time to diagnosis from initial presentation was on average 7.4 weeks (range of 0.3-40 weeks). Approximately 78% of the cases (57 of 73) had biopsies with histology that confirmed Aspergillus fungal morphology, and 30/73 (41%) had diagnostic fungal cultures. The majority of the cases received monotherapy with intravenous (IV) amphotericin B (36 patients; 49%) and IV voriconazole (19 patients; 26%), with a combination of the 2 or more antifungal agents being used in 11 patients (15%). Forty patients (55%) showed signs of clinical improvement with treatment, while 33 (45%) patients did not experience any improvement or continued to deteriorate, and 23 (32%) died in the course of their reported follow-up. Conclusions: The present cases illustrate well the challenge in the diagnosis and treatment of OAS due to invasive sinus aspergillosis. Our review and analysis of 73 cases support the notion that a high index of suspicion leading to early biopsy with histology and fungal culture is paramount for diagnosis. Early empiric antifungal treatment and debridement can potentially reduce morbidity and mortality.

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Aspergillosis: An Unusual Case of Orbital Apex Syndrome

Cited by 2 publications

References 4 publications

Orbital apex syndrome secondary to aspergilloma masquerading as a paranasal sinus tumor

Orbital apex syndrome secondary to aspergilloma masquerading as a paranasal sinus tumor

Orbital Apex Syndrome Secondary to Invasive Aspergillus Infection: A Case Series and Literature Review

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