xtractio Case report and K view of the litera Dolores Martinez, MD, DDS,’ Miguel Burgue2.0, Yv~D,~ Gabriel Forteza, MD,c Mercedes Martin, MD, DDS,d and Ignacio Sierra, MD,e Madrid, Spain, and Palma de Mallorca, Spain HOSPITAL
“LA
PAZ”
Paranasal sinus aspergillosis has usually been considered a rare disease, but it is seen more frequently in both immunocompromised and immunocompetent persons. Invasion may reach the sinuses via the nose or from the mouth after dental procedures. Even though the infection is usually limited to one or more sinuses, it may in certain cases extend to vascular or intracranial structures with a fatal outcome. In these cases, aggressive treatment is justified. We report a case of maxillary sinus aspergillosis that developed after dental extraction. Attention should be given to this possibility even after an apparently uncomplicated dental extraction. (ORAL SURG ORAL MED
ORAL PATHQL
199&74:466-g)
A spergillus, a fungus of the Ascomycetes group, is widely distributed in nature. It can be isolated from many different sources-stored grain, grass, dead leaves, soil, refrigerator walls, wet paint, building materials. Conidia are inhaled regularly and can often be isolated from open air. Aspergillus hyphae are identified in tissuesas filamentous structures about 2 to 4 ym in diameter, septate, and branching at 45-degree angles. This fungus must be differentiated from Mucor, which forms broader hyphae, usually nonseptate, that branch dichotomously at 90-degree angles. Although hundreds of different speciesare known, only a few thermotolerant strains are pathogenic for human beings-A. jiimigatus, A. flaws, A. niger. The infection is not considered to be transmissible among human beings or from animals to human beings.
Even though
inhalation
of spores is common,
diseaseis very rare, and, in its pulmonary invasive aFifth-year Resident in Oral and Maxillofacial Surgery, La Paz. bOral and Maxillofacial Surgeon, La Paz. “Oral and Maxillofacial Surgeon, Hospital San Dureta, Palma de
Mallorca. dFourth-year
Resident
in Oral and Maxillofacial
Vhief, Department of Oral and Maxiilofacial 7113136983 466
Surgery,
La Paz.
Surgery, La Paz.
form, is almost entirely limited to immunosuppressed hosts. However, Aspergillus sinusitis is not exceptional in the normal host, in whom it represents the most common form of paranasal fungal infection.’ We present a case of Aspergillus sinusitis that appeared after a dental extraction. CASE REPORT
A 35-year-oldwomanwasadmittedto our hospitalwith a 2-monthhistory of fever (38.5’ C, 101’ F) andthrobbing pain in the right side of the face. She had received several courses of antibioticswithout improvement.Shedeniedany
history of seriousillnessand did not smokeor drink. One monthbeforethe beginningof the symptoms,her first right uppermolarhadbeenextractedbecauseof pulp disease; the procedurehad beenuneventful. On physicalexamination,right facial swellingwas evident with marked tenderness.A greenishgelatinousright nasaldischargewasnoticed.The patient’sgeneralcondition wasgoodexcept for a fever (101’ F.). Her vision wasnormalin botheyes,aswasintrinsicandextrinsicmobility. The results of hemograms, blood glucose and tests for creatinine, liver function and electrolytes were within normal limits, as was a coagulation screening. The chest x-ray film was normal. A Waters projection of the facial sinuses showed opacification of the right maxillary sinus with a small area of increased density. The possibility of a metallic foreign body accidentally introduced at the time of the dental extraction was considered, but the image was not confirmed
Volume Number
74 4
Maxillary
aspergillosis
467
in a tomographic study which clearly showed erosion of the medial wall of the right maxillary sinus. Complete removal of the maxillary sinus mucosa was carried out with the Caldwell-Luc procedure. Microscopic examination of the surgical specimen disclosed wide areas of mucosal necrosis with invasion of the corion by septate hyphae with branching at 45 degrees (Fig. l), which were identified as an Aspergillus species. No cultures were performed. Amphotericin B was administered up to a total dose of 2 gm. Pain and swelling disappeared after the first 10 days of treatment. A follow-up 6 months later did not disclose any signs of disease. DISCUSSION Stammberger et a1.2 quote Schubert as the first to describe aspergiilosis of the nose and paranasal sinuses in 1885. This condition is infrequently seen worldwide in either immunocompromised or previously healthy patients, except for the northern Sudan, where it is endemic. It primarily affects the maxillary and less often the ethmoid and sphenoid sinuses.3 As in the case of the lung, Hora4 diff’erentiates between nasal sinus invasive and noninvasive forms. More recently a fulminant foam and an allergic form have been added. The noninvasive form usually appears in immunocompetent patients who have facial pain and a gelatinous nasal discharge. Long-term treatment with broad-spectrum antibiotics, facial trauma, and nasal obstruction are accepted predisposing factors.5> 6 Legent et aL7 observed several cases of sinu.s aspergillosis after perforation of the maxillary sinus during endodontic treatment. In most of their cases, zinc oxide-eugenol was found floating free in the maxillary sinus. Merle recently de Foer et al.8 have proposed two different pathways of infection: aerogenie and dentogenic. The invasive form appears as a mass that affects the paranasal sinuses, orbit, and, occasionally, the overlying skin. It may invade central nervous system structures. It is usually, but not always, seen in immunocompromised patients. Differential diagnosis with neoplasia, mucocele, mucormycosis, and granulomatous diseases is mandatory.3, g, lo The fulminant form appears only in immunocompromised patients. The inferior turbinate and septal mucosa are covered with crusts and show marked hypoesthesia. The paranasal sinuses and the central nervous system are rapidly invaded, and facial swelling, ocular proptosis, and chemosis become evident. The infection disseminates to lungs, liver, spleen, and other distant organs lhrough vascular invasion. The most frequent cause of death is thrombosis of the middle cerebral artery. It should be differentiated from mucormycosis aLnd midline granuloma.” 1
Fig. I. Aspergillus fumigatus hyphae. (Hematoxylineosin stain; original magnification X450.)
The allergic form is seen in young adults with a history of atopy (usually asthma and nasal polyps). The clinical pattern is that of a chronic sinusitis with nasal obstruction. More than one or even all the paranasal sinuses are simultaneously involved. This form represents Coombs type I and II immunologic reactions to Aspergillus antigens. In 85% of the cases there is an increased titer of total IgE, and a positive cutaneous immediate response to Aspergillus is present in 65% of the cases.12, l3 A diagnosis of sinus aspergillosis can be suspected clinically and should be confirmed histologically. Species identification requires a positive culture. Radiologic manifestations have been well described by Stammberger.2 The noninvasive form appears in its early stages as a soft-tissue lesion with blurred margins and represents the mycelar mass. Further enlargement of the mass leads to an increased opacity. Very dense concretions are seen in nearly one half of the cases; these radiopaque structures consist mainly of tertiary calcium phosphate (apatite), as well as smaller quantities of calcium sulfate and heavy metal salts deposited in the necrotic areas of the mycelium.
Martinez
et al.
Differential diagnosis should include metallic foreign bodies or misplaced dental fillings.2~ l4 The invasive and fulminant forms usually show accompanying erosion of bone. The allergic form typically involves more than one sinus. Computerized tomography is more sensitive than standard radiography or pluridirectional tomography in the depiction of the calcium or metal components of the fungal infection. Magnetic resonance imaging is even more sensitive than computerized tomography in the identification of fungal concretions. Decreased signal intensity on T2-weighted magnetic resonance images has been described as characteristic of mycetomas. l5 For both noninvasive and invasive forms, surgical debridement and drainage of the affected sinus through transoral antrotomy or lateral rhinotomy is the recommended treatment. In cases of bone, vascular, or meningeal invasion, a more aggressive approach is indicated, such as radical maxillectomy with orbital exenteration when there is invasion of the central retinal artery or other ocular structures. In invasive and fulminant forms, amphotericin B should be administered in addition to the surgical procedure.9-“T i5 Universal agreement on the use of amphotericin B in the noninvasive forms has not been reached. Optimal treatment of the allergic form is yet to be established. Waxman et a1.i3 administer systemic steroids after debridement in recurrent cases, and inhaled steroids in slowly evolving forms of allergic sinus aspergillosis. In conclusion, even though sinus aspergillosis is considered a rare condition, its frequency is on the increase, probably because of widespread use of antibiotics and immunodepressant drugs. The clinician should be aware of this possibility, especially after dental manipulation or extraction, and confirm the diagnosis by means of a biopsy. This would allow early treatment and a better prognosis. Treatment is surgical in nearly all cases. In the invasive and fulminant forms, antifungal drugs should be added. In the allergic form, systemic or inhaled steroids are indicated, with or without surgical treatment.
~RAi!hRG@tAifViED
ORAi PATMOL October 1992
1. Glass RBJ, Hertzanu Y, Mendelsohn DB, Pasen J. Paranasai sinus aspergillosis: a case report with computed tomogram findings. J Laryngoi Otol 1984;98:199-205. 2. Stammberger H, Jakse R, Beaufort F. Aspergillosis of the paranasal sinuses. X-ray diagnosis, histopathology and clinical aspects. Ann Otol Rhino1 Laryngol 1984;93:251-6. 3. Vu VL, Wagner GE, Shadomy S. Sinoorbital aspergillosis treated with combination antifungal therapy. JAMA 19813; 244:814-5. 4. Hora JF. Primary aspergillosis of the paranasal sinus and associated areas. Laryngoscope 1965;75:768-73. 5. McGuirt WF, Harili JA. Paranasal sinus aspergillosis. Laryngoscope 1979;89:1563-8. 6. Axelsson H, Carlsoo B, Weibling I, Winbiad B. Aspergillosis of the maxillary sinus: clinical and histopathological features of four cases and review of the literature. Acta Otolaryngol 1978;86:303-8. I. Legent F, Beauvillain C, Mercier J, Hoffmann B, Wesoluch M. Rapports entre l’aspergillosis sinusienne, les rhinolithes primitifs et la pathologie dentaire. Ann. d’otolaryngol. 1982; 99:541-5. 8. De Foer C, Fossion E, Vaillant JM. Sinus aspergillosis. J Craniomaxillofac Surg 1990;18:33-40. 9. Yumoto E. Kitani S. Okamura H. Yananihara N. Sinoorbital aspergillosis associated with total ophthalmoplegia. Laryngoscope 1985;95:190-2. 10. Sarti EJ, Blaugrund SM, Lin PT, Camins MB. Paranasal disease with intracranial extension: aspergillosis versus malignancy. Laryngoscope 1988;98:632-5. 11. McGill TJ, Simpson 6, Healy GB. Fulminant aspergillosis of the nose and paranasal sinuses: a new clinical entity. Laryngoscope 1980;90:748-54. 12. Katzenstein ALA, Sale SR, Greenberger PA. Allergic aspergillosis sinusitis: a newly recognized form of sinusitis. J. Allergy Clin Immunol 1983;72:89-93. 13. Waxman JE, Spector JG, Sale SR, Katzenstein ALA. Allergic aspergillosis sinusitis: concepts in diagnosis and treatment of a new clinical entity. Laryngoscope 1987;97:261-5. 14. Kopp W, Fotter R, Steiner H, Beaufort F, Stammberger H. Aspergillosis of the paranasal sinuses. Radiology 1985; 156:715-6. 15. Zinreich SJ, Kennedy DW, Malat J, et al. Fungal sinusitis; diagnosis with CT and MR imaging. Radiology 19&8;169:43944.
Reprint requests. Dolores Martinez, MD, Arapiles u. 15 28015 Madrid Spain
DDS
“LA
PAZ”
Paranasal sinus aspergillosis has usually been considered a rare disease, but it is seen more frequently in both immunocompromised and immunocompetent persons. Invasion may reach the sinuses via the nose or from the mouth after dental procedures. Even though the infection is usually limited to one or more sinuses, it may in certain cases extend to vascular or intracranial structures with a fatal outcome. In these cases, aggressive treatment is justified. We report a case of maxillary sinus aspergillosis that developed after dental extraction. Attention should be given to this possibility even after an apparently uncomplicated dental extraction. (ORAL SURG ORAL MED
ORAL PATHQL
199&74:466-g)
A spergillus, a fungus of the Ascomycetes group, is widely distributed in nature. It can be isolated from many different sources-stored grain, grass, dead leaves, soil, refrigerator walls, wet paint, building materials. Conidia are inhaled regularly and can often be isolated from open air. Aspergillus hyphae are identified in tissuesas filamentous structures about 2 to 4 ym in diameter, septate, and branching at 45-degree angles. This fungus must be differentiated from Mucor, which forms broader hyphae, usually nonseptate, that branch dichotomously at 90-degree angles. Although hundreds of different speciesare known, only a few thermotolerant strains are pathogenic for human beings-A. jiimigatus, A. flaws, A. niger. The infection is not considered to be transmissible among human beings or from animals to human beings.
Even though
inhalation
of spores is common,
diseaseis very rare, and, in its pulmonary invasive aFifth-year Resident in Oral and Maxillofacial Surgery, La Paz. bOral and Maxillofacial Surgeon, La Paz. “Oral and Maxillofacial Surgeon, Hospital San Dureta, Palma de
Mallorca. dFourth-year
Resident
in Oral and Maxillofacial
Vhief, Department of Oral and Maxiilofacial 7113136983 466
Surgery,
La Paz.
Surgery, La Paz.
form, is almost entirely limited to immunosuppressed hosts. However, Aspergillus sinusitis is not exceptional in the normal host, in whom it represents the most common form of paranasal fungal infection.’ We present a case of Aspergillus sinusitis that appeared after a dental extraction. CASE REPORT
A 35-year-oldwomanwasadmittedto our hospitalwith a 2-monthhistory of fever (38.5’ C, 101’ F) andthrobbing pain in the right side of the face. She had received several courses of antibioticswithout improvement.Shedeniedany
history of seriousillnessand did not smokeor drink. One monthbeforethe beginningof the symptoms,her first right uppermolarhadbeenextractedbecauseof pulp disease; the procedurehad beenuneventful. On physicalexamination,right facial swellingwas evident with marked tenderness.A greenishgelatinousright nasaldischargewasnoticed.The patient’sgeneralcondition wasgoodexcept for a fever (101’ F.). Her vision wasnormalin botheyes,aswasintrinsicandextrinsicmobility. The results of hemograms, blood glucose and tests for creatinine, liver function and electrolytes were within normal limits, as was a coagulation screening. The chest x-ray film was normal. A Waters projection of the facial sinuses showed opacification of the right maxillary sinus with a small area of increased density. The possibility of a metallic foreign body accidentally introduced at the time of the dental extraction was considered, but the image was not confirmed
Volume Number
74 4
Maxillary
aspergillosis
467
in a tomographic study which clearly showed erosion of the medial wall of the right maxillary sinus. Complete removal of the maxillary sinus mucosa was carried out with the Caldwell-Luc procedure. Microscopic examination of the surgical specimen disclosed wide areas of mucosal necrosis with invasion of the corion by septate hyphae with branching at 45 degrees (Fig. l), which were identified as an Aspergillus species. No cultures were performed. Amphotericin B was administered up to a total dose of 2 gm. Pain and swelling disappeared after the first 10 days of treatment. A follow-up 6 months later did not disclose any signs of disease. DISCUSSION Stammberger et a1.2 quote Schubert as the first to describe aspergiilosis of the nose and paranasal sinuses in 1885. This condition is infrequently seen worldwide in either immunocompromised or previously healthy patients, except for the northern Sudan, where it is endemic. It primarily affects the maxillary and less often the ethmoid and sphenoid sinuses.3 As in the case of the lung, Hora4 diff’erentiates between nasal sinus invasive and noninvasive forms. More recently a fulminant foam and an allergic form have been added. The noninvasive form usually appears in immunocompetent patients who have facial pain and a gelatinous nasal discharge. Long-term treatment with broad-spectrum antibiotics, facial trauma, and nasal obstruction are accepted predisposing factors.5> 6 Legent et aL7 observed several cases of sinu.s aspergillosis after perforation of the maxillary sinus during endodontic treatment. In most of their cases, zinc oxide-eugenol was found floating free in the maxillary sinus. Merle recently de Foer et al.8 have proposed two different pathways of infection: aerogenie and dentogenic. The invasive form appears as a mass that affects the paranasal sinuses, orbit, and, occasionally, the overlying skin. It may invade central nervous system structures. It is usually, but not always, seen in immunocompromised patients. Differential diagnosis with neoplasia, mucocele, mucormycosis, and granulomatous diseases is mandatory.3, g, lo The fulminant form appears only in immunocompromised patients. The inferior turbinate and septal mucosa are covered with crusts and show marked hypoesthesia. The paranasal sinuses and the central nervous system are rapidly invaded, and facial swelling, ocular proptosis, and chemosis become evident. The infection disseminates to lungs, liver, spleen, and other distant organs lhrough vascular invasion. The most frequent cause of death is thrombosis of the middle cerebral artery. It should be differentiated from mucormycosis aLnd midline granuloma.” 1
Fig. I. Aspergillus fumigatus hyphae. (Hematoxylineosin stain; original magnification X450.)
The allergic form is seen in young adults with a history of atopy (usually asthma and nasal polyps). The clinical pattern is that of a chronic sinusitis with nasal obstruction. More than one or even all the paranasal sinuses are simultaneously involved. This form represents Coombs type I and II immunologic reactions to Aspergillus antigens. In 85% of the cases there is an increased titer of total IgE, and a positive cutaneous immediate response to Aspergillus is present in 65% of the cases.12, l3 A diagnosis of sinus aspergillosis can be suspected clinically and should be confirmed histologically. Species identification requires a positive culture. Radiologic manifestations have been well described by Stammberger.2 The noninvasive form appears in its early stages as a soft-tissue lesion with blurred margins and represents the mycelar mass. Further enlargement of the mass leads to an increased opacity. Very dense concretions are seen in nearly one half of the cases; these radiopaque structures consist mainly of tertiary calcium phosphate (apatite), as well as smaller quantities of calcium sulfate and heavy metal salts deposited in the necrotic areas of the mycelium.
Martinez
et al.
Differential diagnosis should include metallic foreign bodies or misplaced dental fillings.2~ l4 The invasive and fulminant forms usually show accompanying erosion of bone. The allergic form typically involves more than one sinus. Computerized tomography is more sensitive than standard radiography or pluridirectional tomography in the depiction of the calcium or metal components of the fungal infection. Magnetic resonance imaging is even more sensitive than computerized tomography in the identification of fungal concretions. Decreased signal intensity on T2-weighted magnetic resonance images has been described as characteristic of mycetomas. l5 For both noninvasive and invasive forms, surgical debridement and drainage of the affected sinus through transoral antrotomy or lateral rhinotomy is the recommended treatment. In cases of bone, vascular, or meningeal invasion, a more aggressive approach is indicated, such as radical maxillectomy with orbital exenteration when there is invasion of the central retinal artery or other ocular structures. In invasive and fulminant forms, amphotericin B should be administered in addition to the surgical procedure.9-“T i5 Universal agreement on the use of amphotericin B in the noninvasive forms has not been reached. Optimal treatment of the allergic form is yet to be established. Waxman et a1.i3 administer systemic steroids after debridement in recurrent cases, and inhaled steroids in slowly evolving forms of allergic sinus aspergillosis. In conclusion, even though sinus aspergillosis is considered a rare condition, its frequency is on the increase, probably because of widespread use of antibiotics and immunodepressant drugs. The clinician should be aware of this possibility, especially after dental manipulation or extraction, and confirm the diagnosis by means of a biopsy. This would allow early treatment and a better prognosis. Treatment is surgical in nearly all cases. In the invasive and fulminant forms, antifungal drugs should be added. In the allergic form, systemic or inhaled steroids are indicated, with or without surgical treatment.
~RAi!hRG@tAifViED
ORAi PATMOL October 1992
1. Glass RBJ, Hertzanu Y, Mendelsohn DB, Pasen J. Paranasai sinus aspergillosis: a case report with computed tomogram findings. J Laryngoi Otol 1984;98:199-205. 2. Stammberger H, Jakse R, Beaufort F. Aspergillosis of the paranasal sinuses. X-ray diagnosis, histopathology and clinical aspects. Ann Otol Rhino1 Laryngol 1984;93:251-6. 3. Vu VL, Wagner GE, Shadomy S. Sinoorbital aspergillosis treated with combination antifungal therapy. JAMA 19813; 244:814-5. 4. Hora JF. Primary aspergillosis of the paranasal sinus and associated areas. Laryngoscope 1965;75:768-73. 5. McGuirt WF, Harili JA. Paranasal sinus aspergillosis. Laryngoscope 1979;89:1563-8. 6. Axelsson H, Carlsoo B, Weibling I, Winbiad B. Aspergillosis of the maxillary sinus: clinical and histopathological features of four cases and review of the literature. Acta Otolaryngol 1978;86:303-8. I. Legent F, Beauvillain C, Mercier J, Hoffmann B, Wesoluch M. Rapports entre l’aspergillosis sinusienne, les rhinolithes primitifs et la pathologie dentaire. Ann. d’otolaryngol. 1982; 99:541-5. 8. De Foer C, Fossion E, Vaillant JM. Sinus aspergillosis. J Craniomaxillofac Surg 1990;18:33-40. 9. Yumoto E. Kitani S. Okamura H. Yananihara N. Sinoorbital aspergillosis associated with total ophthalmoplegia. Laryngoscope 1985;95:190-2. 10. Sarti EJ, Blaugrund SM, Lin PT, Camins MB. Paranasal disease with intracranial extension: aspergillosis versus malignancy. Laryngoscope 1988;98:632-5. 11. McGill TJ, Simpson 6, Healy GB. Fulminant aspergillosis of the nose and paranasal sinuses: a new clinical entity. Laryngoscope 1980;90:748-54. 12. Katzenstein ALA, Sale SR, Greenberger PA. Allergic aspergillosis sinusitis: a newly recognized form of sinusitis. J. Allergy Clin Immunol 1983;72:89-93. 13. Waxman JE, Spector JG, Sale SR, Katzenstein ALA. Allergic aspergillosis sinusitis: concepts in diagnosis and treatment of a new clinical entity. Laryngoscope 1987;97:261-5. 14. Kopp W, Fotter R, Steiner H, Beaufort F, Stammberger H. Aspergillosis of the paranasal sinuses. Radiology 1985; 156:715-6. 15. Zinreich SJ, Kennedy DW, Malat J, et al. Fungal sinusitis; diagnosis with CT and MR imaging. Radiology 19&8;169:43944.
Reprint requests. Dolores Martinez, MD, Arapiles u. 15 28015 Madrid Spain
DDS
