| A forty-year-old man underwent an allogeneic BMT from his HLA identical sister. GvHD prophylaxis was done with cyclosporine (CyA), methotrexate and prednisone (PDN). On day 90 extensive GvHD was noted and higher doses of immunosuppressive drugs alternating CyA with PDN were initiated. Patient's follow-up was complicated by intermittent episodes of leukopenia and monthly episodes of sinusitis or pneumonia. One year after BMT, the patient developed hoarseness and nasal voice. No etiologic agent could be identified on a biopsy sample of the vocal chord. Upon tapering the doses of immunosuppressive drugs, the patient had worsening of chronic GvHD and was reintroduced on high doses of cyclosporine alternating with prednisone on day 550. Three months later, GvHD remained out of control and the patient was started on azathioprine. On day 700, hoarseness and nasal voice recurred. Another biopsy of the left vocal chord failed to demonstrate infection. Episodes of sinusitis became more frequent and azathioprine was withheld 3 months after it was started. One month later, the patient had bloody nasal discharge and surgical drainage of maxillary sinuses was performed. Histopathology showed hyphae and cultures grew scedosporium apiospermum. itraconazole 800 mg/day was initiated. The patient developed progressive respiratory failure and died 15 days later. ( info) |
| Invasive aspergillosis and fulminant aspergillosis are rarities with a high mortality. In the literature there is no patient surviving an extended invasive aspergillosis of the paranasal sinuses and skull base after failure of operative intervention and of postoperative amphotericin b therapy. We report a complete remission of an invasive, partially fulminant aspergillosis. After an incomplete removal of the mycotic mass, we started postoperative drug therapy with amphotericin b. Under this treatment, the mycosis progressed. Additionally, the patient developed severe side effects, so that the treatment was interrupted. At this moment, we started a combined antimycotic drug therapy with liposomal amphotericin b and itraconazole. Within 10 weeks, clinically and radiologically, there was complete remission. The patient died 63 weeks after this treatment, due to a fulminant bacterial pneumonia. Postmortem histologic examination showed no aspergillosis in the skull base, paranasal sinuses, or lung. ( info) |
3/653. Ophthalmic manifestations of allergic fungal sinusitis. PURPOSE: To highlight allergic fungal sinusitis as a cause of ophthalmic and sinus problems by identifying the profile of the patient with allergic fungal sinusitis and presenting a successful treatment approach. methods: Six consecutive cases of patients with ophthalmic manifestations of allergic fungal sinusitis were reviewed. Ophthalmic findings, sinus involvement, mycology, immune response, imaging studies, and treatment were examined. The characteristics of this patient group with ophthalmic manifestations of allergic fungal sinusitis were compared with those of the general group of patients with allergic fungal sinusitis. RESULTS: All six patients had proptosis. One had symptomatic diplopia and one had visual loss. Imaging studies, fungal characterization, and immune profiles were similar to the reported allergic fungal sinusitis population. After treatment there was no recurrence of ophthalmic or sinus symptoms at a mean follow-up of 34 months (range, 8 to 48 months). There were no complications of treatment. CONCLUSIONS: Initial diagnosis of allergic fungal sinusitis requires suspicion on the part of the ophthalmologist. Proptosis is the most common ophthalmic sign. Differentiation from invasive forms of fungal sinus disease is crucial, because systemic antifungal medication and extensive surgical tissue debridement are not required in allergic fungal sinusitis. Treatment consists of extirpation of the allergic mucin and fungus, sinus aeration, and systemic and topical corticosteroids. ( info) |
| PURPOSE: To report successful treatment of ocular infection caused by the microsporidium encephalitozoon cuniculi in a person with acquired immunodeficiency syndrome (AIDS) and nasal and paranasal sinus infection. METHOD: Case report. RESULTS: Microsporidial infection in a person with AIDS and with chronic sinusitis and keratoconjunctivitis was diagnosed by Weber modified trichrome stain and transmission electron microscopy. Symptoms completely resolved with itraconazole treatment (200 mg/day for 8 weeks) after albendazole therapy (400 mg/day for 6 weeks) was unsuccessful. CONCLUSION: itraconazole can be recommended in ocular, nasal, and paranasal sinus infection caused by E. cuniculi parasites when treatment with albendazole fails. ( info) |
5/653. Allergic fungal sinusitis. A report of two cases with diagnosis by intraoperative aspiration cytology. BACKGROUND: Allergic fungal sinusitis (AFS) is a newly recognized form of sinusitis characterized by opacification of the paranasal sinuses by "allergic mucin" (AM) admixed with scattered fungal organisms. AM consists of necrotic, or partially necrotic, eosinophils and Charcot-Leyden crystals suspended in lakes of laminated, mucinous material. AFS is characterized by the absence of bone or soft tissue invasion, purulent exudate or granulomatous inflammation. Clinically, it is important to differentiate AFS from both acute invasive fungal sinusitis and noninvasive mycetoma because the three diseases are treated with different modalities and have different prognoses. Although the radiologic features of AFS are often characteristic, occasionally it may be difficult to exclude neoplasia. CASES: Two cases of AFS, in which intraoperative diagnosis was made on the basis of the presence of both AM and fungal organisms, are reported. CONCLUSION: Cytologic diagnosis of AFS can be made from intraoperative sinus aspirates from the presence of AM and fungal elements. AM and fungi provide presumptive evidence for a noninvasive, allergic fungal disease and can help reassure clinicians intraoperatively and guide clinical management. ( info) |
6/653. Endoscopic sinus surgery in the management of mucormycosis. This is a report of the use of endoscopic sinus surgery in the management of three patients diagnosed with rhino-orbital or rhino-orbito-cerebral mucormycosis. A retrospective review was performed of the clinical examinations and imaging studies of three patients who underwent endoscopic sinus surgery as part of their therapy for mucormycosis. In addition to endoscopic surgery, all patients had aggressive control of underlying risk factors (diabetes mellitus, immunosuppression) and prolonged intravenous amphotericin b therapy. All three patients survived and avoided orbital exenteration. In selected patients with rhino-orbito-cerebral mucormycosis, endoscopic techniques can play a valuable role in diagnosis and management. ( info) |
7/653. Non-Hodgkin's lymphoma of the lacrimal sac and concomitant pansinusitis. Paranasal sinus disease has an established propensity to breach anatomical barriers and present with orbital clinical features. Lacrimal sac lymphomas on the other hand are rare, usually present in males in the sixth decade and 50 per cent of cases have systemic lymphoma/leukaemia. Atypical presentations of lymphomas at this and other sites are reported in patients with acquired immunodeficiency syndrome (AIDS). The clinical features, investigations and treatment of a young human immunodeficiency virus (hiv) sero-negative woman with a non-Hodgkin's lymphoma localized to the lacrimal sac and presenting with a concomitant ipsilateral pansinusitis is described. The importance of submitting surgical material for pathological investigation is stressed. ( info) |
8/653. A proposal for a staging system for hyperplastic rhinosinusitis based on the presence or absence of intranasal polyposis. This study of 30 patients with hyperplastic rhinosinusitis reveals that the major determinant of the success or failure of conservative therapy is the presence or absence of intranasal polyposis. When polyposis is confirmed both clinically and radiographically, surgical intervention appears to be the rational way to handle most cases. When intranasal polyps are not present, an aggressive medical regimen is called for, one that includes a short-term, low-dose oral corticosteroid and an antibiotic, along with periodic radiographic assessment. Regardless of how much disease is seen on computed tomography, if it does not also detect intranasal polyps or masses, the potential for clearing with conservative therapy is extremely high. ( info) |
| Mucosa-associated lymphoid tissue (MALT) is a specialized form of lymphoid tissue that may be acquired at sites in response to chronic inflammation. Most low-grade, B-cell, non-Hodgkin lymphomas that occur at extranodal sites derive from acquired MALT. Confusing and overlapping terms have been used to describe these lymphomas, but immunohistochemical advances now allow more precise subtyping. Our review of the literature yielded only 2 previous reports of sinonasal MALT-derived lymphoma, and we report an additional case in a patient with a history of chronic sinusitis. Current developmental theories of MALT-derived lymphomas are discussed. In addition, we review the clinical, histologic, and immunophenotypic features of MALT-derived lymphomas. ( info) |
| lemierre syndrome seldom follows an episode of pharyngotonsillitis. Characteristically, it is comprised of septic thrombosis of the internal jugular vein and bacteremia, leading to lung emboli and metastatic abscess formation. We describe lemierre syndrome that complicates an acute mastoiditis, with considerations regarding its pathogenesis and management. Despite its sporadic occurrence, awareness of lemierre syndrome is important, since early recognition reduces both the morbidity and mortality associated with it. ( info) |