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3 Van Gompel for Website Copyright © 2009, Barrow Neurological Institute Neurosurgical Management of Bipolaris-Specific Skull Base Allergic Fungal Sinusitis: Diagnostic Criteria and Outcome ver the past decade, the incidence † Jamie J. Van Gompel, MD Oof fungal infections has increased Scott D. Wait, MD dramatically. Fungal paranasal sinus dis- ‡ Stephen W. Coons, MD ease, a large component of fungal in- William L. White, MD fections, is common and is defined by the host and the host’s response to the Skull base allergic fungal sinusitis is a rare but important neurosurgical entity that fungus. Invasive fungal sinusitis occurs can be mistaken for a skull base tumor during preoperative assessment. Due to in immunocompromised hosts and can the significantly variable clinical presentation of and preoperative evaluation for manifest as acute, fulminant, and life- this disease, clinicians are often surprised when the diagnosis becomes appar- threatening or as a more manageable ent during surgery or thereafter. However, SBAFS must be differentiated from chronic/granulomatous indolent form. malignancy and invasive fungal disease because this allergic disease does not However, the hallmark of invasive dis- require aggressive, complete resection or potentially toxic antifungal medica- ease is the presence of large amounts of tions. We report six cases of SBAFS to illustrate the neurosurgical management the fungal element in the associated pa- of this rare disease. thology without immune containment. Key Words: allergic fungal sinusitis, Bipolaris, skull base In contrast, noninvasive fungal si- nusitis occurs in immunocompetent in- dividuals. It exists in two forms, both characterized by the host’s response to the fungal agent. With the first, myce- toma, the host’s immune response is in- adequate and the fungal agent proliferates as an encapsulated mass. The second, AFS, the subject of this report, occurs when the host provides an overly exu- berant immune response to the antigens of the fungus. Skull base allergic fungal sinusitis, a term first introduced by Kinsella et al., denotes the specific condition in which AFS extends intracranially.10 Ten to 20% of patients with AFS have an in- tracranial extension.13 Once SBAFS is encountered, neurosurgical interven- tion is required. Because of their variable presentation and rarity, these Abbreviations Used: AFS, allergic fungal sinusitis; CT, computed to- lesions are seldom expected preopera- 15 mography; GMS, Gomori methenamine silver; MR, magnetic resonance; tively. Consequently, SBAFS is often SBAFS, skull base allergic fungal sinusitis mistaken for a malignancy,11,12,18,27 and patients may receive overly aggressive Divisions of Neurological Surgery and ‡Neuropathology, Barrow Neurological Institute, St. therapy. Because SBAFS is noninva- Joseph’s Hospital and Medical Center, Phoenix, Arizona sive, surgical debulkment is standard Current Address: †Saint Marys Hospital at the Mayo Clinic, Rochester, Minnesota therapy; radical resection or toxic med- 14 BARROW QUARTERLY • Vol. 24, No. 1 • 2008 Van Gompel et al: Neurosurgical Management of Bipolaris-Specific Skull Base Allergic Fungal Sinusitis: Diagnostic Criteria and Outcome Table 1. Clinical Summary of Six Patients with Bipolaris SBAFS Case Age/ Presenting Presenting Medical Surgery Bacterial Neurological Sex Symptoms† Signs History Isolate‡ F/U 1 31/M HA, OD vision loss, OD blindness/RAPD, NP, asthma, TNTS Staphylococcus resolved OS blurring Dysconjugate gaze chronic sinusitis 2 25/F OS vision loss OS vision loss NP, allergies, SLTS Staphylococcus resolved chronic sinusitis 3 20/F HA, N/V OD proptosis and NP, asthma TNTS Staphylococcus resolved vision loss 4 36/M Chronic nasal CN 6 paresis NP, allergies, SLTS§ Haemophilus resolved drainage chronic sinusitis 5 75/F Tremor, falling, HA, Head and upper NP, allergies TNTS Staphylococcus resolved decreasing memory extremity tremor 6 19/M OD proptosis and Diplopia on left NP, allergies, BCC Staphylococcus resolved blurring lateral upward gaze asthma †No patient had presenting symptoms consistent with pituitary malfunction. ‡All cases cultured Bipolaris heavily; bacterial isolate represents coincident culture. §Complicated by aseptic meningitis. F/U = follow-up, HA = headache, N/V = nausea and vomit- ing, OD = right eye, OS = left eye, RAPD = relative afferent pupil defect (nonreactive pupil), NP = nasal polyps, CN = cranial nerve, TNTS = transnasal transsphenoidal, SLTS = sublabial transsphenoidal, BCC = bicoronal craniotomy. ical regimens are unnecessary. We pre- quent sign was ocular defect. All pa- weighted MR images lacked lesional or sent six patients with Bipolaris-specific tients had a history of atopy, including perilesional hyperintensity, suggesting no SBAFS to highlight the neurosurgical polyps and at least one other indicator inflammation. All but one lesion was iso- management of this rare clinical entity. of atopy. intense to brain parenchyma; the excep- tion was hypointense (Table 2). Four cases showed intense uptake on gadolin- Bipolaris-Specific AFS ium-enhanced MR imaging, and an- Between January 1998 and August Clinical Materials and other lesion was considered ring en- 2004, 28 cases of culture-proven Bipo- Methods hancing. There was no gadolinium laris-specific AFS were treated at our in- enhancement in the remaining case. In Bipolaris-Specific SBAFS stitution. There were 20 men and 8 all cases CT showed increased attenua- The records of six patients (3 men, 3 women (mean age, 30 years; range, 13 to tion and bony destruction with expan- women; mean age, 34 years; age-range, 80 years). Three patients had an intra- sion of its associated sinus or sella without 19 to 75 years) who underwent resec- cranial extension consistent with SBAFS evidence of hyperostosis. The location of tion with pathologic verification of AFS (invasion rate, 10.7%). Of these 28 cases, the pituitary in relation to the tumor var- and culture-proven Bipolaris from Janu- only 1 had fungal elements identified ied across the cases. ary 1989 to August 2004 were analyzed on a KOH preparation. Eleven had retrospectively. The criteria for patho- concomitant positive cultures for bac- logic verification were allergic mucin- teria. Three patients who underwent Treatment containing eosinophils, Charcot-Ley- surgery were not included in the mi- Five cases consisted of a primary den crystals, cellular debris, and sparse crobiology database because they pre- sphenoidal extension and were treated hyphae. To evaluate the invasive nature dated its establishment. through a transsphenoidal approach of this disease, all cases of Bipolaris-spe- (Table 1). A tumor that originated from cific AFS were identified by querying the frontal sinus and extended into the our microbiology database from Janu- Results anterior cranial fossa was approached ary 1998 to August 2004. through a bicoronal craniotomy. In all Most patients became symptomat- Radiology cases a coincident positive culture of bac- ic with visual complaints or headache Bipolaris-specific SBAFS lesions were terial isolate (nasopharyngeal flora) was of gradually increasing intensity (Table isointense on T1-weighted MR images also treated (Table 1). Postoperatively, 1). On examination, the most fre- relative to brain tissue (Table 2). T2- one patient was diagnosed with aseptic BARROW QUARTERLY • Vol. 24, No. 1 • 2008 15 Van Gompel et al: Neurosurgical Management of Bipolaris-Specific Skull Base Allergic Fungal Sinusitis: Diagnostic Criteria and Outcome Table 2. Bipolaris SBAFS Imaging Characteristics Case MRI† Gadolinium Attenuation† CT Bone Pituitary T1 T2 Uptake Destruction Location‡ 1 Isointense Isointense Intense Increased + Posterior 2 Isointense Isointense Intense Increased + Elevated 3 Isointense Isointense Ring enhancing Increased + Posterior 4 Isointense Isointense Intense Increased + Anterior 5 Isointense Isointense No uptake Increased + Posterior 6 Isointense Hypointense Intense Increased + None †Relative to brain parenchyma. ‡Relative to mass and sella. meningitis. The resulting transient re- atopy, nasal polyposis, chronic sinusitis Moreover, involvement of the optic nerve current sixth cranial nerve palsy resolved with eight previous sinus surgeries, and suggested chiasmopathy and involvement with treatment. Whether this complica- childhood asthma. of the perichiasmal segment (Fig. 3). tion was related to surgery is unclear. After surgical decompression neurologic Physical Examination Treatment symptoms resolved in all cases (range of On neurologic examination the pa- The patient began steroid therapy and follow-up, 2 months to 15 years). All pa- tient was alert and oriented. His right his vision subsequently improved. He tients received postoperative steroids from pupil was nonreactive to light; his left then underwent frameless stereotactic 2 weeks to 4 months. Three patients also pupil was miotic but briskly reactive. Vi- transnasal-transsphenoidal debulking of received 2 weeks of antifungal therapy. sion was full to confrontation in the left the lesion. A firm white mass intermin- There were no recurrences. eye. Extraocular movements were intact gled with fibrous tissue, which had erod- bilaterally; however, gaze was dyscon- ed the planum sphenoidale and anterior jugate. Evaluation of pituitary function and basal sellar floors, was encountered. was unremarkable. Its adherence to the sellar diaphragm and Illustrative Case frontal fossa dura made its removal diffi- Patient History Radiological Examination cult. An intraoperative frozen-section A 31-year-old African-American
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