ABSTRACT
CASE: A 44-year-old male with past medical history of type II insulindependent diabetes mellitus (DM) and end stage liver disease (ESLD) due to alcohol use and nonalcoholic fatty liver disease (NAFLD) presented with one week of left-sided retroorbital headache and diplopia. Two weeks prior, the patient tested positive for COVID-19 and initially his severe headache was attributed to this diagnosis. On hospital presentation the patient was found to have ophthalmoplegia, ptosis and diminished sensation in the CN V1 distribution on the left. The patient was in diabetic ketoacidosis (DKA) with glucose of 686, venous blood gas of 7.32/29/15 and serum anion gap of 17. Contrasted orbital and maxillofacial CT showed complete opacification of the left sphenoid sinus and CT angiography/venography of the head were negative for venous sinus thrombosis. MRI of the brain showed left optic nerve ischemia and left frontal lobe cerebritis without abscess. Bedside nasal endoscopy with ENT showed purulent, fuzzy white debris bilaterally concerning for fungal sinusitis. He was taken urgently to the operating room and was found to have angioinvasive fungal sinusitis with cultures growing Lichthemia corymbifera, a fungus in the Mucor family. In addition to treatment with IV insulin and fluids for DKA, the patient was given amphotericin B and posaconazole;however, surgical intervention was deemed too high risk and futile in the setting of patient's comorbidities. IMPACT/DISCUSSION: Mucormycosis is a fungal infection that typically involves the sinuses, orbits and the central nervous system (CNS). Infection of the sinuses manifests with fever, sinus congestion/pain and headache, but can rapidly progress to involve the orbits, leading to vision changes, and the CNS, leading to encephalopathy. Other structures that can be involved include the cavernous sinus, leading to palsies of cranial nerves III-VI. Known risk factors for mucormycosis include DM, especially in patients with DKA, glucocorticoid treatment, immunosuppression and deferoxamine use. Urgent histopathologic diagnosis, initiation of intravenous antifungal agents (amphotericin B) and surgical intervention with ENT, ideally prior to extension beyond the sinuses, are fundamental to decreasing mortality, which is as high as 62%. There have been numerous case reports of mucormycosis in patients with COVID-19, particularly from India. Many of these patients were prescribed glucocorticoids as part of the COVID-19 treatment pathway or had underlying DM. Additional research is needed into the association between COVID-19 and invasive mucormycosis. CONCLUSION: In patients with poorly controlled DM or immunosuppression presenting with severe headache, sinus pain, and/or neurologic changes, mucormycosis must be considered, as it is a fatal entity requiring urgent surgical intervention and initiation of antifungal agents. Patients with COVID-19 infection may be at increased risk for mucormycosis, especially in those with underlying DM or on glucocorticoids.
ABSTRACT
Curvularia species are dematiaceous filamentous fungi that can cause a variety of infections in both immunocompetent and immunocompromised hosts. We present 2 cases of severely immunosuppressed patients with acute invasive fungal sinusitis due to Curvularia species. Both patients had a history of hematologic malignancy with refractory disease and prolonged neutropenia. They presented with facial and sinus pain, which prompted maxillofacial computed tomography that showed acute sinusitis. Subsequently, they underwent nasal endoscopy with a biopsy that revealed a definitive diagnosis of invasive fungal sinusitis. Dematiaceous fungi are responsible for most fungal sinusitis cases, with Curvularia being one of the most common species isolated. Generally, invasive fungal rhinosinusitis may follow a relatively innocuous and nonspecific course. In addition, fungal infections may complicate chronic allergic sinusitis. Computed tomography scan is the first imaging modality of choice, and magnetic resonance imaging has a role in prognostication in acute invasive fungal rhinosinusitis. Endoscopic sinus surgery with biopsy yields a definitive diagnosis and is therapeutic. Management typically includes a combination of surgery and antifungal agents. Severe neutropenia is a significant risk factor for infection and is associated with poor outcomes. Aggressive surgical debridement, combined with antifungal therapy, should be emphasized in leukemic patients despite their prolonged neutropenia and bleeding tendency.