Criteria for the diagnosis of sinus mycetoma Richard D. deShazo, MD, Margaret O'Brien, MD, Kimberle Chapin, MD, Maria Soto-Aguilar, MD, Ronnie Swain, MD, Michael Lyons, MD, W. Carter Bryars, Jr., MD, and Steven Alsip, MD Mobile, Ala. Recent improvements in the treatment options for fungal disease of the sinuses make rapid distinction between the syndromes of invasive and noninvasive sinusitis necessary. On the basis of the extensive review of the literature detailed here, we developed case-finding criteria for the noninvasive syndrome of sinus mycetoma. Using these criteria, we identi- fied 20 cases of sinus mycetoma in the medical literature and compared findings in these patients to those of five patients with mycetoma evaluated in our clinics. Like those in the literature, our patients appeared immunocompetent and were often first seen for evaluation of symptoms other than those usually associated with sinusitis. Two were first seen with a new-onset seizure disorder and one with nasal obstruction alone. Patients from both groups more commonly had myce- toma in their maxillary sinuses, and fungus failed to grow from the cheesy, grey-green, hyphae-rich material removed at the time of surgery. Clinical features in our five patients, which are not noted in published reports of sinus mycetoma, included frequent atopy, nasal polyps, calcification within the sinus on computed tomography, and an excellent response to surgical treatment. Serendipitously, one patient had both allergic fungal sinusitis and a mycetoma in the same sinus. On the basis of this experience, we have modified our case- finding criteria into proposed diagnostic criteria for sinus mycetoma. The elements of these criteria are designed to: (1) exclude patients with invasive fungal sinusitis and (2) differ- entiate sinus mycetoma from other forms of noninvasive fun- gal sinusitis on the basis of specific histopathologic findings. This study and the criteria presented reflect our view that sinus mycetoma represents a distinct but not necessarily iso- lated element in the spectrum of noninvasive fungal disease of the sinuses. (J Allergy Clin Immunol 1997;99:475-85.) Key words: Sinus mycetoma, fungal sinusitis, computed tomographic scans, continuouspositive airwaypressure Although fungal diseases of the sinuses were first reported in the medical literature over 200 years ago, there is still uncertainty as to their appropriate classifi- cation? -4 This uncertainty has led to confusion in report- ing and dilemmas in the clinic.5 A precise clinical classification of these conditions is more than an aca- demic exercise, because invasive sinus disease may lead to death, and misdiagnosis of noninvasive disease may From the Division of Allergy and Immunology, Departments of Medi- cine and Pediatrics, the Departmentof Pathology,and Departmentof Surgery, Collegeof Medicine,University of South Alabama,Mobile. Receivedfor publication Mar. 26, 1996;revisedOct. 11, 1996; accepted for publication Oct. 14, 1996. Reprint requests: Richard D. deShazo, MD, University of South Ala- bama, Collegeof Medicine,Departmentof Medicine,2451 Fillingim St., Mastin400A,Mobile,AL 36617. Copyright© 1997 by Mosby-Year Book, Inc. 0091-6749/97 $5.00 + 0 1/1/78559 Abbreviations used AFS: Allergic fungal sinusitis ANA: Antinuclear antibody CT: Computed tomography lead to therapeutic misadventure. Newer surgical tech- niques and less toxic antifungal antibiotics allow rapid diagnosis and more effective treatment of fungal sinusitis and have increased the importance of accurate diagno- sis. Breaching of the sinus mucosa by fungal elements with fungal growth into the submucosa and underlying bone has been used as the key feature to distinguish invasive from noninvasive fungal sinusitis. 3, 6 Even so, apparently noninvasive fungal sinusitis has evolved into invasive disease. 7-9 This has led some authors to propose that fungal sinusitis is not a group of discrete diseases but a series of conditions that overlap. 4 In this context, "destructive, noninvasive fungal sinusitis" has been pro- posed as a separate entity. 4 For the last several years, our group has been actively studying patients with fungal disease of the sinuses, including the syndromes of allergic fungal sinusitis (AFS) and fungal mycetoma? ° In the process of these studies and a review of the literature, we found that clinicopathologic criteria for the diagnosis of these two conditions have neither been proposed nor validated. Absence of diagnostic criteria has resulted in an inability to discriminate between invasive and noninvasive dis- ease in many of the previous reports. 11 In this study we reviewed the available literature on fungal mycetoma including clinical and histopathologic descriptions of this disease and approaches to therapy. On the basis of this review, we developed clinicopatho- logically based case-finding criteria for sinus mycetoma. These criteria were used to identify five previously unreported cases of sinus mycetoma in our patient population. Histopathologic studies of specimens ob- tained from these patients showed a varied pattern of mucosal inflammation in response to the fungus and indicated that mycetoma may coexist with other forms of fungal sinusitis, including AFS. This experience suggests that the syndrome of sinus mycetoma may be precisely identified by using the criteria proposed and that sinus mycetoma represents a distinctive but not necessarily isolated element in the spectrum of noninvasive fungal disease of the sinuses. 475
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