Endoscopic treatment of allergic fungal sinusitis

Endoscopic treatment of allergic fungai sinusitis HUMA A. QLIRAISHI,MD, and HASSAN H. RAMADAN, MD, FACS,Morgantown, West Virginia

Fungal sinusitis has been seen increasingly in immunocompetent individuals. The literature has stressed the importance of diagnosing these cases. Surgical debridement is the treatment of choice. Six immunocompetent individuals with allergic fungal sinusitis were seen at West Virginia University between January 1992 and December 1993. After adequate evaluation, endoscopic surgical debridement was performed. After surgery they were treated with systemic or topical steroids and oral antibiotics for several weeks. Two patients required a second procedure within a year. Four of our six patients had positive fungal cultures for Aspergillus or Curvularia. None of our patients had evidence of invasive disease on histopathology, and systemic antifungal therapy was not required. Patient course and response will be discussed in detail along with a brief review of allergic iungal sinusitis. ( O t o l a r y n g o l Head Neck Surg 1997;117:2%34.)

S i n c e Katzenstein et al.'s 1 initial description of allergic aspergillosis of the sinuses, there haw~ been multiple reports of fungal sinusitis in immunocompetent individuals. Furthermore, multiple dematiaceous fungi, such as BipoIaris, Alternaria, CurvuIaria, and Exserohilum, have been isolated from affected individuals. 2 This has led to the more general term allergic fungal sinusitis (AFS). 3 To date, the literature has stressed the importance of recognizing this entity. Surgical debridement and aeration of the affected sinus is generally accepted as the treatment of choice. Endoscopic sinus surgery has been advocated, even though the results of this type of management have not been reported in the English literature. A recent French study retrospectively reviewed 40 cases of fungal sinusitis treated endoscopically, with a mean follow-up of 13 months. Thirty-eight patients were found to have Aspergillusfumigatus, and 39 were free of recurrence during the follow-up period. 4 Systemic antifungal therapy, which was once advocated, is no longer deemed necessary unless there is pathologic evidence of invasive disease. Systemic

From the Department of Otolaryngology-Head and Neck Surgery, West Virginia University. Presented at the Annual Meeting of the American Academy of Otolaryngology-Headand Neck Surgery, New Orleans, La., Sept. 17-20, 1995. Reprint requests: Hassan H. Ramadan, MD, FACS, Department of Otolaryngology-Headand Neck Surgery,WestVirginia University, P.O. Box 9200, Morgantown, WV 26506-9200. Copyright © 1997 by the American Academy of OtolaryngologyHead and Neck SurgeryFoundation, Inc. 0194-5998/97/55.00 + 0 2311173735

Fig. 1. CT scan revealing left maxillary sinus mass extending into the ethmoid sinuses with erosion of the medial orbital wall, Note areas of focal affenuation suggestive of fungal process.

steroids, which have been the mainstay of treatment for the related disorder allergic bronchopulmonary aspergillosis, remain controversial. We review our experience with six patients with AFS at West Virginia University who were treated with endoscopic sinus surgery with or without steroids. We also present an illustrative case.

CASE REPORT A 23-year-old woman sought treatment for a 5-month history of pressure and pain in her left cheek and progressive left-sided proptosis. Her medical history was significant for 29

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30 QURAISHI and RAMADAN

Fig. 2. Coronal TI MRI image of patient in Fig. 1.

allergies but she had no history of asthma or aspirin sensitivity. On physical examination the patient was noted to have a large polypoid mass filling the left nasal cavity. CT scan and MRI were obtained (Figs. 1, 2, and 3). The patient was given preoperative prednisone and underwent a left endoscopic maxillary antrostomy and total ethmoidectomy. Intraoperatively, thick, pasty greenish brown material, inspissated mucus, and polypoid mucosal changes were observed (Figs. 4 and 5). Cultures were positive for a Curvularia species and Klebsiella pneumoniae. After surgery the patient began antibiotic therapy, an aggressive regimen of saline lavage, and aerosolized beclomethasone use. A follow-up CT scan 2 months later revealed persistent opacification of the left frontal sinus with total aeration of the left maxillary sinus. A revision endoscopic procedure was performed to open up the nasofrontal region. Fungal cultures were negative. She was given a course of oral steroids, 60 mg prednisone tapered over 2 weeks, and then prednisone 5 mg daily for another 2 months. She has been taking aerosolized beclomethasone for the past 2 years. She remains symptom free with no evidence of mucosal disease on nasal endoscopy. METHODS A N D MATERIAL

Between January 1992 and January 1995, six patients with AFS were treated with endoscopic debridement and aeration of their sinuses. All patients underwent bilateral middle meatal maxillary antrostomies and total ethmoidectomy. After surgery, packs were placed in the middle meatus for 1 week. Patient ages ranged between 3 and 70 years, with a mean of 37 years. There were two male and four female patients (Table 1). At presentation all of our patients had syrup-

toms of chronic sinusitis refractory to medical management except for the above patient, who had a nasal mass on CT. Medical management included multiple courses of antibiotics and short-term courses of aerosolized beclomethasone. All adult patients had multiple unilateral sinus involvement, whereas our pediatric patients had disease limited to the maxillary sinuses. The diagnosis of AFS was based on the presence of fungal hyphae on Gomori's methenamine stain accompanied by allergic mucin, characterized by an eosinophilic infiltrate (Fig. 4) and positive fungal cultures. 5-1° RESULTS

All six patients underwent endoscopic debridement while under general anesthesia. Three patients were noted to have thick, greenish yellow, pasty material. Two had cheesy white debris, and one had yellow concretions. All but one of our patients had evidence of fungal hyphae on Gomori's methenamine staio. In addition, four of our patients had positive fungal cultures. On histopathologic analysis, no evidence was found of tissue invasion, and none of our patients required systemic antifungal therapy. After surgery two patients received systemic steroids, 60 mg prednisone orally tapered over 8 weeks. Two patients had recurrent symptoms and required revision procedures. The four remaining patients are symptom free and have no evidence of nasal polyposis or mucosal changes in the sinuses. Follow-up has ranged from 4 months to 3 years after surgery.

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QURAISHI and RAMADAN 31

Fig. 3, Comparison of T1 ( A ) and T2 (B) images. Note that fungat sinusitis appears hypointense on T1 i m a g e and even more hypointense on T2 image,

DISCUSSION AFS has been reported to account for up to 7% of all operative casesJ ° Typically it presents in a younger patient population, although as our cases illustrate, it has been reported in all age groups. 1°'13 Almost all

patients are atopic and have nasal polyps and multiple unilateral sinus involvement. In addition, roughly 50% will have associated asthma, and up to 25% will have aspirin sensitivityJ ° The most common presenting symptoms are nasal obstruction caused by polyps and

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Toble 1. Patient characteristics Patient no

Age (yr)

Sex

Presentation

1

70

F

Chronic sinusitis

Right maxillary & ethmoid opacification

2

4

F

Chronic sinusitis

3

23

F

4

65

5

3

CT findings

56

Followup

Pathology

Culture

Treatment

Cheesy white material

Allergic mucin with hyphae

No growth

FESS, symptoms recurred, revision FESS

4 mo

Bilateral maxillary opacification

Yellow-brown material

Allergic mucin with hyphae

No growth

FESS

2 yr

Left proptosis

Left maxillary & ethmoid opacification

Yellow-brown material

Allergic mucin

Curvularia

FESS, symptoms recurred, revision FESS, steroids

2 yr

F

Chronic sinusitis

Left maxillary & ethmoid opacification

Yellow-brown material

Allergic mucin with hyphae

Aspergillus

FESS

3 yr

M

Chronic sinusitis

Right maxillary opacification

Yellow concretions

Allergic mucin with hyphae

Aspergillus

FESS

2 yr

Chronic sinusitis

Left maxillary & ethmoid opacification

Cheesy material

Allergic mucin with hyphae

Aspergillus

FESS, steroids

1 yr

i

6

Operative findings

M

i

u

FESS,Functionalendoscopic sinus surgery.

local pain. Proptosis and intracranial extension may also occur in long-standing cases] On endoscopy, these patients have been noted to have inspissated "peanut butter" like mucin, although two of our patients had thick, cheesy, white debris. The CT scan (Fig. 1) of this patient demonstrates areas of focal attenuation suggestive of fungal disease or a neoplasm. MRI has proved to be a more specific modality. Fungal disease appears hypointense on T 1 imaging and even more hypointense on T 2 imaging (Fig. 3). By contrast, neoplasms and bacterial infections have increased signal intensityJ 4 Even though MRI is more specific, all of our patients underwent CT scanning for assessment of bony landmarks. Bony destruction, as demonstrated in Fig. 1, has been reported in up to 60% of casesJ 5 This is a result of pressure necrosis and is indicative of the chronicity of the disease rather than invasiveness. Recently the dematiaceous fungi, particularly Bipolaris (Dreschlera), Alternaria, and Curvularia, have been identified in AFS more commonly than Aspergillus] °J6 Interestingly, three of our four patients with culture-positive sinusitis grew Aspergillus. The hallmark of AFS is "allergic mucin" characterized by eosinophilia and Charcot-Leyden crystals (Fig. 4) and fungal hyphae on Gomori's methenamine and periodic acid-Sct~f stains. Five of six patients had microscopic evidence of fungal

disease. The only patient lacking hyphae on pathologic analysis had positive cultures for Curvularia. There have been multiple reports of patients who have responded to debridement and aeration through an external approach. 11-21All six of our patients were treated endoscopically with middle meatal maxillary antrostomies and total ethmoidectomy on the affected side. By contrast, a recent European study advocated both middle and inferior meatal maxillary antrostomies and catheter placement in the inferior antrostomy for lavage for 1 week after surgery.4 Fottr of our six patients remain symptom free after their initial endoscopic procedure. Two patients experienced recurrence within months of their primary procedures. We believe that this is the result of inadequate initial debridement. Both patients underwent revision endoscopic procedures and remained symptom free for 4 months and 2 years, respectively. After surgery all patients began receiving intranasal steroids. Two of our patients began taking systemic steroids. Even though this treatment method remains controversial, we believe there is a role for high-dose systemic steroids for several weeks in patients with AFS. CONCLUSION

We report on six patients with AFS. All six of our patients underwent endoscopic surgical management of their disease. Four of six patients required only one pro-

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QURAISHI and RAMADAN 33

Volume 117 Number 1

Fig. 4. "Allergic mucin" with eosinophils.

Fig. 5. Nasal mucosa with inflammatory and eosinophilic infiltrate.

cedure. Two patients required a revision procedure. We attribute primary treatment failure in these two patients to inadequate initial debridement. We c o n c l u d e that in selected cases endoscopic sinus surgery is appropriate and successful in the m a n a g e m e n t of AFS. We thank Kimberly ReVeal and Beth Ann Dlugos for helping to prepare this manuscript.

REFERENCES i. Katzensteil~ALA, Sale SR, Greenbeyer PA. Allergic aspergitlus sinusitis: a newly recognized form_ of sinusitis. J Allergy Clin Immunol 1983;72:89-93. 2. Lawson W; Blitzer A. Fungal infections of the nose and paranasal sinuses, part II. Otolaryngol Clin North Am 1993;26:1037-68. 3. Robson JMB, Benn RAV, Hogan PG, et al. Allergic fungal sinusitis presenting as a paranasal sinus tumor. Aust N Z J Med 1989;19:351-3.

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4. de Gaudemar I, Ebbo D, Leconte F, et al. Les mycoses du sinus maxillaire: a props de 40 cas. Ann Otol Laryngol (Paris) 1993;110:198-202. 5. Katzenstein ALA, Sale SR, Greenbeyer PA. Pathologic findings in allergic Aspergillus sinusitis. Am J Surg Pathol 1983;7:439-43. 6. Bent JP, Kuhn FA. Diagnosis of allergic fungal sinusitis. Otolaryngol Head Neck Surg 1994;111:580-8. 7. Corey JR Delsaphe KG, Ferguson BJ. Allergic fungal sinusitis: allergic, infectious, or both? Otolaryngol Head Neck Surg 1995;113:110-9. 8. AllphinAL, Strauss M, Abdul Karim FW. Allergic fungal sinusitis: problems in diagnosis and treatment. Laryngoscope 1991;101:815-20. 9. Manning SC, Schafer SD, Close LG, et al. Culture positive allergic fungal sinusitis. Arch Otolaryngol Head Neck Surg 1991;117:174-8. 10. Cody DT, Neel B, Feirerro JA, et al. Allergic fungal sinusitis: the Mayo clinic experience. 1994;104:1074-9. 11. Waxman JE, Spector GJ, Sale SR. Allergic aspergillosis sinusitis: concepts in diagnosis and treatment of a new clinical entity. Laryngoscope 1987;97:261-6. 12. Ence BK, Gourley DS, Jorgensen NL, et al. Allergic fungal sinusitis. Am J Rhinol 1990;4:169-78.

13. Manning SC, Viutch F, Weinberg AG, et al. Allergic aspergillosis: a newly recognized form of sinusitis in the pediatric population. Laryngoscope 1989;99:681-5. 14. Zinreich SJ, Kennedy DW, Malat J, et al. Fungal sinusitis: diagnosis with CT and MR imaging. Radiology 1988;169:439-44. 15. Handly GH, Visscher DW, Katzenstein ALA, et al. Bone erosion in allergic fungal sinusitis. Am J Rhinol 1990;4:149-53. 16. Zieske LA, Kopke RD, Hamill R. Dematiaceous fungal sinusitis. Otolaryngol Head Neck Surg 1991; 105:567-77. 17. Daghistani KJ, Jamal TS, Zaher S, et al. Allergic Aspergillus sinusitis with proptosis. J Laryngol Otol 1992; 106:799-803. 18. Nishioka G, Schwartz G, Renaldi M, et al. Fungal maxillary sinusitis caused by Curvularia lunata. Arch Ototaryngol Head Neck Surg 1987;113:665-6. 19. Bartynski JM, McCaffrey TV, Evanjelos E Allergic fnngal sinusitis secondary to dematiaceous fungi: Curvularia and Alternaria. Otolaryngol Head Neck Surg 1990; 103:32-9. 20. Pingree TF, Holt GR, Otto RA, et al. Bipolaris caused fungal sinusitis. Otolaryngol Head Neck Surg 1992;106:302-5. 21. Kelleyshovich SM, Wetmore SJ. Curvularia/Dreschlera sinusitis. Laryngoscope 1990;100:932-7.

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