Basidiomycetous (mushroom) infection of the maxillary sinus PETER CATALANO, MD, WILLIAM LAWSON, MD, DDS. EDWARD BOTTONE, PhD, and JEFFREY LEBENGER, MD.
New York. New York
C h r o n i c sinusitis involving the maxillary antrum is a common problem confronting the otolaryngologist. Usual pathogens include gram-positive and gramnegative organisms, as well as anaerobes. Fungal maxillary sinusitis is usually encountered in the immunocompromised host; however, recent reports show an increased incidence among the immunocompetent. We have recently treated a case of chronic right maxillary sinusitis in an otherwise healthy woman who had an expansile lesion of the right antrum. Although two species of Aspergillus were initially cultured from nasal smears, the Basidiomycetous (mushroom) fungus was the only pathogen identified from operative cultures and pathology specimens. To our knowledge, this is only the third case of rnaxillary sinusitis in which the Basidiomycetous fungus has been identified as a primary pathogen. Case history, management, and review of the literature follows.
’
CASE REPORT
A 35-year-old woman was referred to the Mount Sinai Medical Center with a history of intermittent midfacial pain, nasal congestion, and associated toothache for the previous 3 months. She had completed several courses of oral antibiotic therapy with only partial symptomatic relief. She denied any history of systemic disease, but related inhalant allergy. She also denied history of facial trauma, previous surgery, distant travel, cocaine, or other drug abuse. On physical examination there was deflection of the nasal septum to the right, with right nasal mucosal congestion. The septum was otherwise intact. The remaining examination of the head and neck was essentially negative. Her hemoglobin was 12.2 gm, hematocrit 35.6%, and white cell count 9600. Differential was essentially normal, with 3% eosinophils. Erythrocyte sedimentation rate was 42. Chest x-ray film was unremarkable. Roentgen examination of the sinuses revealed opacification of the right antrum and clouding of the right ethmoid without
From the Department of Otolaryngology and Microbiology, Mount Sinai Medical Center. Submitted for publication April 24, 1989; revision received Aug. 14, 1989; accepted Aug. 15, 1989. Reprint requests: William Lawson, MD, DDS, Department of Otolaryngology, Mount Sinai Medical Center, 100th St. and Fifth Ave., New York, NY 10029. 2314116076
an air-fluid level. An attempt at right antral lavage produced some mucopus, and cultures were taken at this time. The woman was placed on trimethoprim and sulfamethoxazole and nasal decongestants that gave her partial symptomatic relief. Follow-up x-ray films showed persistent opacification of the right antrum. Results of tuberculin skin tests at this time were negative; the anergy panel was normal. The patient refused HIV testing. Nasal1antral cultures identified two species of Aspergillus: Aspergillus niger and Aspergillus glaucus. A CT scan of the paranasal sinuses revealed a soft tissue density expanding the right maxillary antrum, with extension into the right ethmoid sinus. There was also some opacification of the right frontal sinus. The sphenoid sinuses were normal. There was no evidence of bone erosion, although medial displacement of the medial wall of the right maxillary antrum was noted. The patient was taken to the operating room and underwent right intranasal ethmoidectomy and Caldwell-Luc procedure with antrostomy. The right antrum was totally filled with polypoid mucosa and a grey-black, gritty material, which packed the sinus and could be seen extending into the nasal cavity. All of this material was carefully removed using a malleable curette and cupped forceps. The surgical contents were cultured in the mycology laboratory where the basidiomycetous fungus, schizophyllum commune, was identified. Bacteriologic cultures grew normal flora. Microscopic study of the specimen showed eosinophilic infiltration of the polypoid nasal mucosa. An acute and chronic inflammatory infiltrate was also present. The patient’s postoperative course was unremarkable. One year later, she remains free of disease.
DISCUSSION
Basidiomycete fungi are well-known and widespread plant pathogens. They attack a wide range of host trees, where the mushroom-shaped basidiocarps are easily identified (Fig. l).’ They are whitish in color when young, but become darker shades of grey at maturity. They are identified by their characteristic hyphae and production of extracellular oxidases. Their pathogenic potential was studied by Greer and bola no^,^ who injected an inoculum of the fungus into the peritoneal cavity of white mice of various ages. Only the youngest mice died mortality approached 50%. When the mice were pretreated with cortisone, mortality increased to nearly 75% in all age groups except adult mice, in which group the mortality remained zero. However, disease was produced in all age
’
183
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184 CaseReports
Fig. 1. Schizophy//umcommune grown in culture from a maxiliary sinus infection.
groups, with or without cortisone. The authors concluded that induced infection follows a course characteristic of opportunistic fungi-infection of low virulence which, when aided by immunosuppression, will lead to death in the youngest hosts. The basidiomycetous fungus has been recovered from the cerebrospinal fluid of a patient with atypical meningitis; from the sputum of a patient with chronic lung disease; and from an ulcerating lesion on the hard palate of a 4-month-old girl with vomiting and dehyd r a t i ~ nA . ~different species of basidiomycetous fungus, Coprinus cinereus, was isolated from the sputum of a patient with chronic obstructive pulmonary disease and from the aortic valves of a patient undergoing aortic valve repla~ement.~" The two previously reported cases* of sinusitis resulting from the basidiomycetous fungus also involved the maxilary antrum. The first case was a healthy 30year-old woman with symptoms of chronic sinusitis refractory to medical therapy, who eventually underwent a Caldwell-Lucprocedure to eradicate the disease. Sinus cultures identified S . commune as the primary pathogen. The second patient was a 75-year-old woman with type I1 diabetes who again had a clinical picture of chronic maxillary sinusitis requiring a Caldwell-Luc procedure to control the disease. The sinus contents were described as an accumulation of black, necrotic debris enveloped by markedly edematous sinus mucosa. Again, S . commune was the only organism grown in
culture from the surgical specimen. Both patients were reportedly free of disease 1 year after surgery. Certain features of basidiomycete fungi infections of the paranasal sinuses are common to the three cases reported. Infection appears to be chronic, with partial relief of symptoms after antibiotic therapy; there is predilection for the maxillary sinus; routine sinus x-ray films show an opacified antrum refractory to lavage; the sinus contents are grey-black, gritty, and completely fill the sinus; the Caldwell-Luc procedure with complete removal of the sinus contents is curative; and immunocompetent hosts are not spared. The small number of reported cases makes the observed female predilection of questionable significance. The absence of bacteriologic growth in all three cases may be secondary to previous treatment with multiple antibiotic regimens, or represent an inability of bacteria to withstand the extracellular oxidases produced by the fungus. The chronic nature of the infection is further supported by the grey-black contents found within the antra, as the fungus darkens as it matures. The present case report and those found on review of the medical literature suggest a role for the basidiomycetous fungus as a primary pathogen in human infection, notably maxillary sinusitis. These cases also suggest the fungus may not be a typical "opportunistic" pathogen, since it has never been isolated from an immunocompromised host. Yet, despite its relatively low virulence, chronic sinusitis secondary to this organism
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Volume 102 Number 2 February 1990
Case Reports 185
is associated with significant morbidity, requiring surgery to completely eradicate the disease. REFERENCES
1. Stevens MH. Primary fungal infections of the paranasal sinuses. Am J Otolaryngol 1981;4:348-57. 2. Kern ME, Uecker FA. Maxillary sinus infection caused by the homobasidiomycetous fungus Schizophyllum commune. J Clin Microbiol 1986;6: 1001-5.
3. Greer DL, Bolanos B. Pathogenic potential of schizophyllum commune isolated from a human case. Sahouraudia 1973;ll: 233-44. 4. Restrepo AD, Greer AD, Robledo M, Osorio 0, Mondragon H. Ulceration of the palate caused by a basidiomycete Schizophyllum comune. Sabouraudia 1973;11:201-4. 5. DeVries GA, Kemp RF, Speller DC. Endocarditis caused by coprinus delicurulus. C R Commun Congr ISHAM 1971;5:185-6. 6. Greer DL, Basidiomycetes as agents of human infections: a review. Mycopathologia 1978;65:133-9.
BOUND VOLUMES AVAILABLE TO SUBSCRIBERS Bound volumes of Otolaryngology-Head and Neck Surgery are available to subscribers (only) for the 1989 issues from the Publisher, at an individual cost of $46.00 ($56.00 international) each for Vols. 100 (January-June) and 101 (July-December). Shipping charges are included. Each bound volume contains subject and author indices and all advertising is removed. Copies are shipped within 60 days after publication of the last issue in the volume. The binding is durable blue buckram with the JOURNAL name, volume number, and year stamped in gold on the spine. Payment must accompany all orders. Contact The C. V. Mosby Company, Circulation Department, 11830 Westline Industrial Drive, St. Louis, MO 631463318, USA; phone (800)325-4177, ext. 351. Subscriptions must be in force to qualify. Bound volumes are not available in place of a regular JOURNAL subscription.
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New York. New York
C h r o n i c sinusitis involving the maxillary antrum is a common problem confronting the otolaryngologist. Usual pathogens include gram-positive and gramnegative organisms, as well as anaerobes. Fungal maxillary sinusitis is usually encountered in the immunocompromised host; however, recent reports show an increased incidence among the immunocompetent. We have recently treated a case of chronic right maxillary sinusitis in an otherwise healthy woman who had an expansile lesion of the right antrum. Although two species of Aspergillus were initially cultured from nasal smears, the Basidiomycetous (mushroom) fungus was the only pathogen identified from operative cultures and pathology specimens. To our knowledge, this is only the third case of rnaxillary sinusitis in which the Basidiomycetous fungus has been identified as a primary pathogen. Case history, management, and review of the literature follows.
’
CASE REPORT
A 35-year-old woman was referred to the Mount Sinai Medical Center with a history of intermittent midfacial pain, nasal congestion, and associated toothache for the previous 3 months. She had completed several courses of oral antibiotic therapy with only partial symptomatic relief. She denied any history of systemic disease, but related inhalant allergy. She also denied history of facial trauma, previous surgery, distant travel, cocaine, or other drug abuse. On physical examination there was deflection of the nasal septum to the right, with right nasal mucosal congestion. The septum was otherwise intact. The remaining examination of the head and neck was essentially negative. Her hemoglobin was 12.2 gm, hematocrit 35.6%, and white cell count 9600. Differential was essentially normal, with 3% eosinophils. Erythrocyte sedimentation rate was 42. Chest x-ray film was unremarkable. Roentgen examination of the sinuses revealed opacification of the right antrum and clouding of the right ethmoid without
From the Department of Otolaryngology and Microbiology, Mount Sinai Medical Center. Submitted for publication April 24, 1989; revision received Aug. 14, 1989; accepted Aug. 15, 1989. Reprint requests: William Lawson, MD, DDS, Department of Otolaryngology, Mount Sinai Medical Center, 100th St. and Fifth Ave., New York, NY 10029. 2314116076
an air-fluid level. An attempt at right antral lavage produced some mucopus, and cultures were taken at this time. The woman was placed on trimethoprim and sulfamethoxazole and nasal decongestants that gave her partial symptomatic relief. Follow-up x-ray films showed persistent opacification of the right antrum. Results of tuberculin skin tests at this time were negative; the anergy panel was normal. The patient refused HIV testing. Nasal1antral cultures identified two species of Aspergillus: Aspergillus niger and Aspergillus glaucus. A CT scan of the paranasal sinuses revealed a soft tissue density expanding the right maxillary antrum, with extension into the right ethmoid sinus. There was also some opacification of the right frontal sinus. The sphenoid sinuses were normal. There was no evidence of bone erosion, although medial displacement of the medial wall of the right maxillary antrum was noted. The patient was taken to the operating room and underwent right intranasal ethmoidectomy and Caldwell-Luc procedure with antrostomy. The right antrum was totally filled with polypoid mucosa and a grey-black, gritty material, which packed the sinus and could be seen extending into the nasal cavity. All of this material was carefully removed using a malleable curette and cupped forceps. The surgical contents were cultured in the mycology laboratory where the basidiomycetous fungus, schizophyllum commune, was identified. Bacteriologic cultures grew normal flora. Microscopic study of the specimen showed eosinophilic infiltration of the polypoid nasal mucosa. An acute and chronic inflammatory infiltrate was also present. The patient’s postoperative course was unremarkable. One year later, she remains free of disease.
DISCUSSION
Basidiomycete fungi are well-known and widespread plant pathogens. They attack a wide range of host trees, where the mushroom-shaped basidiocarps are easily identified (Fig. l).’ They are whitish in color when young, but become darker shades of grey at maturity. They are identified by their characteristic hyphae and production of extracellular oxidases. Their pathogenic potential was studied by Greer and bola no^,^ who injected an inoculum of the fungus into the peritoneal cavity of white mice of various ages. Only the youngest mice died mortality approached 50%. When the mice were pretreated with cortisone, mortality increased to nearly 75% in all age groups except adult mice, in which group the mortality remained zero. However, disease was produced in all age
’
183
Downloaded from oto.sagepub.com at University of Sussex Library on June 8, 2016
OtolaryngologyHead and Neck Surgery
184 CaseReports
Fig. 1. Schizophy//umcommune grown in culture from a maxiliary sinus infection.
groups, with or without cortisone. The authors concluded that induced infection follows a course characteristic of opportunistic fungi-infection of low virulence which, when aided by immunosuppression, will lead to death in the youngest hosts. The basidiomycetous fungus has been recovered from the cerebrospinal fluid of a patient with atypical meningitis; from the sputum of a patient with chronic lung disease; and from an ulcerating lesion on the hard palate of a 4-month-old girl with vomiting and dehyd r a t i ~ nA . ~different species of basidiomycetous fungus, Coprinus cinereus, was isolated from the sputum of a patient with chronic obstructive pulmonary disease and from the aortic valves of a patient undergoing aortic valve repla~ement.~" The two previously reported cases* of sinusitis resulting from the basidiomycetous fungus also involved the maxilary antrum. The first case was a healthy 30year-old woman with symptoms of chronic sinusitis refractory to medical therapy, who eventually underwent a Caldwell-Lucprocedure to eradicate the disease. Sinus cultures identified S . commune as the primary pathogen. The second patient was a 75-year-old woman with type I1 diabetes who again had a clinical picture of chronic maxillary sinusitis requiring a Caldwell-Luc procedure to control the disease. The sinus contents were described as an accumulation of black, necrotic debris enveloped by markedly edematous sinus mucosa. Again, S . commune was the only organism grown in
culture from the surgical specimen. Both patients were reportedly free of disease 1 year after surgery. Certain features of basidiomycete fungi infections of the paranasal sinuses are common to the three cases reported. Infection appears to be chronic, with partial relief of symptoms after antibiotic therapy; there is predilection for the maxillary sinus; routine sinus x-ray films show an opacified antrum refractory to lavage; the sinus contents are grey-black, gritty, and completely fill the sinus; the Caldwell-Luc procedure with complete removal of the sinus contents is curative; and immunocompetent hosts are not spared. The small number of reported cases makes the observed female predilection of questionable significance. The absence of bacteriologic growth in all three cases may be secondary to previous treatment with multiple antibiotic regimens, or represent an inability of bacteria to withstand the extracellular oxidases produced by the fungus. The chronic nature of the infection is further supported by the grey-black contents found within the antra, as the fungus darkens as it matures. The present case report and those found on review of the medical literature suggest a role for the basidiomycetous fungus as a primary pathogen in human infection, notably maxillary sinusitis. These cases also suggest the fungus may not be a typical "opportunistic" pathogen, since it has never been isolated from an immunocompromised host. Yet, despite its relatively low virulence, chronic sinusitis secondary to this organism
Downloaded from oto.sagepub.com at University of Sussex Library on June 8, 2016
Volume 102 Number 2 February 1990
Case Reports 185
is associated with significant morbidity, requiring surgery to completely eradicate the disease. REFERENCES
1. Stevens MH. Primary fungal infections of the paranasal sinuses. Am J Otolaryngol 1981;4:348-57. 2. Kern ME, Uecker FA. Maxillary sinus infection caused by the homobasidiomycetous fungus Schizophyllum commune. J Clin Microbiol 1986;6: 1001-5.
3. Greer DL, Bolanos B. Pathogenic potential of schizophyllum commune isolated from a human case. Sahouraudia 1973;ll: 233-44. 4. Restrepo AD, Greer AD, Robledo M, Osorio 0, Mondragon H. Ulceration of the palate caused by a basidiomycete Schizophyllum comune. Sabouraudia 1973;11:201-4. 5. DeVries GA, Kemp RF, Speller DC. Endocarditis caused by coprinus delicurulus. C R Commun Congr ISHAM 1971;5:185-6. 6. Greer DL, Basidiomycetes as agents of human infections: a review. Mycopathologia 1978;65:133-9.
BOUND VOLUMES AVAILABLE TO SUBSCRIBERS Bound volumes of Otolaryngology-Head and Neck Surgery are available to subscribers (only) for the 1989 issues from the Publisher, at an individual cost of $46.00 ($56.00 international) each for Vols. 100 (January-June) and 101 (July-December). Shipping charges are included. Each bound volume contains subject and author indices and all advertising is removed. Copies are shipped within 60 days after publication of the last issue in the volume. The binding is durable blue buckram with the JOURNAL name, volume number, and year stamped in gold on the spine. Payment must accompany all orders. Contact The C. V. Mosby Company, Circulation Department, 11830 Westline Industrial Drive, St. Louis, MO 631463318, USA; phone (800)325-4177, ext. 351. Subscriptions must be in force to qualify. Bound volumes are not available in place of a regular JOURNAL subscription.
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