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Revista Española de Cirugía Oral y Maxilofacial

On-line version ISSN 2173-9161Print version ISSN 1130-0558

Rev Esp Cirug Oral y Maxilofac vol.28 n.2 Barcelona Mar./Apr. 2006

 

PÁGINA DEL RESIDENTE

 

What should the diagnosis be?

¿Cuál es su diagnóstico?

 

 

Female, 35 years old with no previous pathological background of interest, requested treatment with osseointegrated implants (OII) in the third quadrant.

On carrying out the orthopantomography (OPG) an image with a metallic density was observed by chance in the right maxillary sinus (Fig. 1). The patient was clinically asymptomatic. The computerized tomography examination once again showed the presence of a metal-dense image in the right maxillary sinus, together with thickening of the mucosa in the sinus. There were no other findings of note (Fig. 2).

Nasal endoscopy was carried out (Fig. 3) which showed polyposis and very viscous mucus in the sinus that was difficult to drain. Samples were taken for pathologic and microbiological studies (Fig. 4).

 


Sinus aspergillosis with a foreign body appearance

Aspergilosis simulando cuerpo extraño intrasinusal

 

 

A. Pradillos Garcés1, J. Gonzalez Lagunas1, X. Martínez Fuster1, J. Mareque Bueno1,
G. Raspall Martín1, H. Umbert Massegur2

1 Servicio de Cirugía Oral Maxilofacial
2 Servicio de ORL Clínica Girona, Girona, España
Hospital Vall d’Hebron, Barcelona, España

Dirección para correspondencia

 

 

Discussion

Following the chance discovery of an image in a maxillary sinus with a metallic density that is suggestive of a foreign body, the following differential diagnosis should be considered; a) the existence of remains of a root in the sinus, b) presence foreign bodies in the sinus from a different source (e.g. pellet), c) amalgam tattoos, d) non-specific calcifications in the sinus and e) allergic fungal sinusitis (AFS).

In this case some of these diagnoses can be ruled out from the start: in the first two situations (a and b) there would be evidence of the maxilla having been manipulated dentally, or of previous trauma not included in the patient’s medical history. In the event of the implantation of amalgam because of trauma (c) there would be previous dental treatment compatible with the diagnosis, in addition to a tattoo visible on the oral mucosa.1 In this case there was no previous use of amalgam and the examination of the oral mucosa proved normal. An intra-sinusal osteoma could be possible (d), although it is a pathology that is more usually observed in the frontal sinuses, and it is rarely found in the maxillary sinuses, so from the beginning this need not be taken into consideration. In this case, the histological study of the mucus obtained by endoscopy showed an abundance of eosinophils, Charcot-Leyden crystals and scattered fungal hyphae (Fig. 4). This directed the case towards a fungal infection with no mucosal or osseous invasion, and it was suggestive of allergic fungal sinusitis (e).

In view of this diagnostic orientation, endoscopic drainage was carried out of the mucosal blockage and the affected sinus was aerated. The treatment was completed with the administration of systemic corticoids, as the complete elimination of hyphae and spores is unlikely even with the best surgery.

Allergic Fungal Sinusitis (AFS) is a non-invasive fungal disease that has traditionally been linked with an IgE-mediated hypersensitivity reaction to fungal antigens in young adults that are immunocompetent, and that suffer from rhinitis or chronic sinusitis and/or recurrent nasal polyposis in spite of medical and/or surgical treatment.2 There is no sex predominance. On occasions the process can become invasive,3 and bone can even be eroded.

It was first described more than a decade ago by Millar and Lamb4,5 and it was called "Allergic aspergillosis of the sinuses" due to the similarity with "Allergic bronchopulmonary aspergillosis" (ABPA). Subsequent studies showed that most cases with this clinical entity were caused by species that were different to Aspergillus and, as a result, it became known as "Allergic Fungal Sinusitis".

The suspected diagnosis is made when adequate antibiotic treatment for chronic sinusitis repeatedly fails, and less frequently, when there is a radiologic finding of a metallic image in the sinuses that appears to be a foreign body6 (in this event high clinical suspicion is required). There are certain predisposing factors that serve as a guide to diagnosis, such as an atopic host, environmental burden of the fungus, or certain local anatomic conditions such as massive polyposis, septal deviation or obstruction of the osteomeatal complex.

If the condition has been progressing for a long time there may even be exophthalmos, diplopia and intracranial invasion (especially in children).

The radiological study will show dotted radiopaque images with a metallic density in plain x-rays and in computed tomography (CAT scan). The CAT scan will show dense heterogeneous masses within the paranasal sinuses, which may or may not affect the bone. When evaluated by magnetic resonance (MR), these tiny masses can be identified as isointense on T1 and hypointense on T2 weighted images.

When confronted by a clinical picture of these characteristics, a nasal endoscopy should be carried out as a diagnostic and therapeutic procedure. The usual findings are of considerable stasis of secretions preventing sinus drainage together with edema and mucosal inflammation.

In 1994 Bent and Kuhn described five diagnostic criteria that are currently the most widely accepted. These include: 1) Type I hypersensitivity confirmed by the patient’s medical history, skin or serological testing, 2) existence of nasal polyposis, 3) characteristic image of hyperdense material on CAT scan, 4) histological evidence of eosinophilic mucin with no fungal invasion into sinus tissue, and 5) positive fungal staining or culture in the material obtained by surgery of the contents of the sinus (the culture is not essential for the diagnosis). 7 However, there are some studies that claim that the role of IgE in the etiology or pathogeny of AFS has not been demonstrated, and that its diagnosis should not be included in Type I hypersensitivity.2

The definitive diagnosis is therefore histological6 given the appearance of a formation of allergic mucin that contains eosinophils, Charcot-Leyden crystals (a breakdown product of the eosinophils) and scattered fungal hyphae. It is a mass that is rich in water, proteins and lipids. It has a variable quantity of calcium, and it has a high content of metal’s such as zinc, lead, silver, copper, iron;8 the latter could be responsible for the typical radiological images described previously. Multiple fungal species have been identified as etiological factors responsible for the initial inflammation: Curvularia, Bipolaris, Alternaria, Exserohilum, and finally Aspergillus, representing some of the AFS cases, but not the majority.

The treatment of choice for AFS consists in sinusal endoscopic surgery9,10 with drainage of compressed mucus and aeration of the affected sinuses by means of an antrostomy by the middle meatus and/or canine fossa.11 It should be kept in mind that even with the best surgical technique, the elimination of all hyphae and spores is unlikely. It is recommended that the treatment be completed with the administration of systemic corticoids and that an endoscopy be included in the subsequent follow-up in order to evaluate possible recurrences, which would be the norm in the absence of adjuvant therapy after the surgery. There is no reason for using toxic anti-fungal therapies such as Amphotericin B for treating non-invasive fungal sinusitis, nor is there evidence of the efficiency of other drugs such as Itraconazole or Fluconazole.

The prognosis in the short term is good, but both the patient and doctor should understand that, given the current treatment, it is a chronic disease.

 

 

Dirección para correspondencia:
Alba Pradillos Garcés
Servicio de Cirugía Maxilofacial Hospital Vall d’Hebron
P Vall d’Hebron 129
08035 Barcelona
Email: 38640apg@comb.es

 

 

References

1. Buchner A. Amalgam tattoo (amalgam pigmentation) of the oral mucosa: clinical manifestations, diagnosis and treatment. Refuat Hapeh Vehashinayim 2004;21:19-22, 96.        [ Links ]

2. Corradini C, Del Ninno M, Schiavino D, Patriarca G, Paludetti G. Allergic fungal sinusitis. A naso-sinusal specific hyperreactivity for an infectious disease? Acta Otorhinolaryngol Ital 2003;23:168-74.        [ Links ]

3. De Foer C, Fossion E,Vaillant JM. Sinus aspergillosis. J Craniomaxillofac Surg 1990;18:33-40.        [ Links ]

4. Millar J, Lamb D. Allergic bronchopulmonary aspergillosis of the maxillary sinuses. Thorax 1981;36:710.        [ Links ]

5. Lamb D, Millar J, Johnston A. Allergic aspergillosis of the paranasal sinuses. J Pathol 1982;137:56.        [ Links ]

6. Guillen Guerrero VS, Aguirre Garcia F, Munoz Herrera A, Santacruz Ruiz S, Blanco Perez P, Perez Liedo C, Sancipriano Hernandez JA. Maxillary sinusitis caused by Aspergillus. Otorrinolaringol Ibero Am 2000;27:67-75.        [ Links ]

7. Bent JP, Kuhn FA. Diagnosis of allergic fungal sinusitis. Otolaryngol Head Neck Surg 1994;111:580-8.        [ Links ]

8. Braun JJ, Bourjat P, Gentine A, Koehl C, Veillon F, Conraux C. Caseous sinusitis. Clinical, x-ray computed, surgical, histopathological, biological, biochemical and myco-bacteriological aspects. Propos of 33 cases. Ann Otolaryngol Chir Cervicofac 1997;114:105-15.        [ Links ]

9. Plaza G, Toledano A, Plaza A, Oliete S, Noriega J, Galindo N. Non-invasive fungal sinusitis: allergic fungal sinusitis and sinusal mycetomas. Acta Otorrinolaringol Esp 2000;5:603-12.        [ Links ]

10. Carrat X, Rebufy M, Chabrol A. Non invasive naso-sinusal aspergillosis. Contribution of endonasal microsurgery. Apropos of 16 cases. Rev Laryngol Otol Rhinol (Bord) 1993;114:177-81.        [ Links ]

11. Chobillon MA, Jankowski R. What are the advantatges of the endoscopic canine fossa approach in treating maxillary sinus aspergillomas? Rhinology 2004;42:230-5.        [ Links ]

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