Defoer 1990

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j. Cranio-Max.-Fac.Surg.

18 (I990) 33

J.Cranio-Max.-Fac. Surg. 18 (1990) 33-40


9 GeorgThiemeVerlagStuttgart 9 New York
Summary
The prevalence of Aspergillus sinusitis is often under-
Sinus Aspergillosis estimated because the vast majority of cases are classi-
fied as "unspecified sinusitis". Two possible aetio-pa-
thogenic mechanisms can be involved in the develop-
Christophe De Foer*, Eric Fossion*, Jean-Marie Vaillant** ment of this fungal infection. Traditionally, the litera-
ture emphasised the "anglophone" hypothesis which is
*Dept. ofOra/and Head and NeckSurgery based on the inhalation of spores. More recently, the
(Head: Prof. E. Fossion,M.D., D.M.D.),
UniversityHospital Leuven,Belgium "french" model, based on oro-sinusal fistula and/or
**Institute for Stomatologyand Maxillo-FacialSurgery the perforation of the maxillary sinus by root canal-
(Director: Prof. J. M. Vaillant.M.D., D.M.D.)
UniversityHospital "Piti6-Salp&ri~re"Paris, France filling material, is believed to explain the majority of
Submitted 22.12.88; accepted 4.4.89 cases in our industrialised environments. Still, neither
model explains the totality of cases and several remain
beyond comprehension. The disease most commonly
presents as a chronic bacterial sinusitis. The process
can however become invasive, thus resembling malig-
Introduction nancy, with eventually a fatal outcome. Doctors and
Aspergillus is an ubiquitous mould which produces dentists should know the possible danger, presented by
sporulating structures. Inhalation of the spores of any of zinc-oxide-eugenol-paste in the sinus. Radical surgery
the possibly pathogenic species (A. Flavus, A. Niger, and is the treatment of choice, since a prolonged conserva-
most frequently A. Fumigatus) must be extremely com- tive approach (antibiotics, corticosteroids) can only
mon. However, unless there is massive inhalation, or an worsen tile prognosis. This paper discusses different
immunosupressed host, disease is rare. Pulmonary Asper- aspects of the disease, and presents 10 cases, observed
gillosis and Aspergillic otomycosis are relatively common, at the University Hospitals of Paris (France) and Leu-
but Sinus Aspergillosis (S. A.) is an uncommon disease. It ven (Belgium).
presents in two forms; a ball of hyphae, may form in a
chronically obstructed sinus (non-invasive S.A), but, much Key words
less commonly, it may spread slowly to the orbit and brain
(invasive S.A). Sinusitis - Aspergillus - Mycotic Infections - Asper-
gillosis
Terminology and Classification
S. A. was first describes some hundred years ago. At first it
w a s often regarded as a site of syphillis, a very common
disease in those days. In 1906 the Frenchman Texier (cited Aetiopathogenic Theories
by Legent et al. 1982) was the first to exclude the syphilli- Legent et al. (1980) presented a pathogenic mechanism in
tic origin, to mention tile possible dental origin and to re- the French literature, which is hardly ever mentioned in
cognise a "pseudo-tumoral" form of S. A. Fifty years later, the Anglo-American reports. This mechanism is based on
Andersen (Andersen and Steserup, 1956) emphasized the the initial colonisation of the maxillary sinus through a (ia-
importance of Texier's remark that systematic bacteriolog- trogenic) bucco-sinusal connection (e.g. post-extraction
ical investigation would probably show an unexpectedly site, periodontal destruction, root canal perforation).
high number of S.A's. In 1965, Hora (1965) published a This "dental" model should give an alternative for the (in
classification (similar to that of Texier) based on a series of the Anglosaxon literature) widespread concept of spore-in-
25 patients in Texas (Table 1). halation as the one possible explanation for sinus colonisa-
Nowadays, most authors refer to Hora's work as tile basic tion (Vaillant et al., 1983; Braun et al., 1987; Ragot et al.,
classification of S. A. We shall do the same in this article; 1988).
even though Texier's terminology seems more logical to us It is true that the "aerogenic" model can explain tile ma-
than that of his American colleague, sixty years later. Te- jority of cases in all today's larger series. One should how-
xier used a more adequate terminology, since terins such ever keep in mind that most of these reports on S.A.'s are
as "light forms" vs. "pseudo-tumoral forms" describe observed-in entirely different atmospherical conditions
more accurately the radiological images one is confronted e.g. tile Sudan (Milose[ and Mahgoub, 1969; Mahgoub,
with in the clinical situation. The Franco-Anglophone lan- 1971; Veress et al., 1973), Saudi-Arabia (Bassiouny et al.,
guage barrier has had other consequences than polemics 1982) or in India (Bahadur et al., 1983), than the ones we
on terminolgy. S.A. is a relatively rare disease, and since are confronted with in \Vestern-Europe. Traditionally
most authors refer to ideas of fellow countrymen w h o S.A. is observed*in subtropical countries, with a warm,
presented (larger) series in the past, different philosophies moist climate, where tile high Asp. spore-count in the dust
have emerged on either side of the barrier. No historical ("sand-storm's" cfr. Milosef and Mahgoub, 1969) contri-
review of the disease has ever been complete, because of butes considerably to the inoculation of the sinus via nose-
lan.guage problems (Stammberger et al., 1984, Robb 1986). inhalation. Moreover, the subtype of Aspergillus found in
lt-'(vas not our goal to make an exact quantification of patients, recruited in those environements, seems to be
S:A. cases in the literature, kVea'nerely wanted to compare more virulent than the subtype cultured from specimens
those different ideas on aetiopathogenesis and check them from patients living in our \Vestern-European urban con-
against our series. ditions.
34 ]. Cranio-Max.-Fac. Surg. 18 (1990) Christophe De Foer et al.

Fig.1 Arrows on this Panoramic radiograph show a foreign body Fig.2 CT-scan image showing the destruction of the medial wall
in the left maxillary sinus. It can be a eugenate fragment, floating in of the maxillary sinus and invasion of the ethmoid sinus. There is a
the sinus after perforation of the sinus mucosa during root canal metal-dense body in the sinus (case 7 - see also Fig. 1).
treatment on 26 (case 7 - see also Fig.2).

Table I Primary Aspergillosis of the paranasal sinuses and asso-


ciated areas (J. F. Hora, Houston, Texas, 1965)

Non-invasive majority of cases


Aspergillosis symptoms of banal chronic sinusitis
increased resistance to usual therapy
x-rays show antrum-opacification, no bone
destruction
irrigation of antrum yields dark, greasy
material, from which different
Asp. species can be cultured
good prognosis after surgery
Invasive rarely seen
Aspergillosis: serious clinical picture
Pain plus slowly growing mass, mimicking a
malignancy
bone destruction on x-ray

Today, only some 400 case-reports of S. A. have been pub-


lished in the world-literature. In Western-Europe most au-
thors report on one or two isolated cases.

Fig.3 Tomographic radiograph indicating destruction of medial


Material and Methods
,maxillary sinus wall with orbital involvement (case 8).
We have reviewed the patient material between 1976 and
1987 in two maxillo-facial centres; tile Institute for Stom-
In Europe however, one must consider tile higher stand- atology and Maxillo-Facial Surgery in Paris (France) - (see
ards of hygiene and the fact that people request medical case~ 1 to 5) and the Department of Oral and Head and
care at a relatively early stage. Thus we are seldom con- Neck Surgery in Leuven (Belgium)- (see cases 6 to 10). At-
fronted with the late, spectacular forms of S.A. (with the tention has been paid to the clinical and radiological pres-
typical "mastic-like" Aspergilloma) that are seen in the Su- entation of the disease, to the anamnestic factors, to tile
dan, India etc. Most S.A.'s in our countries are therefore peroperativejindings, and to histopathological and bacter-
regarded as banal, bacterial sinusitis and treated as such at iological examination of the collected material, in order to
an early stage. Systematic investigation of the cnretted ma- compare these details with the above-mentioned classifica-
terial is seldom performed. Routine pathology screening tion of Hora (1965) and to both aetiopathogenic theories.
would probably reveal a much higher incidence of S.A:
than is generally thought (Legent et al., 1982). In a recent Case Reports
American study by Zinreich et al. (1988), routine patholo- Case 1: 31-year-old caucasian woman, dental implant in
gy screening revealed 25"fungal sinusitises out of a total location 25, extending into the left maxillary sinus. Im-
number of 293 patients operated on for chronic sinu- plant removed after 6 months and patient kept on anti-
sitis. biotics and non-steroidal auti-inflammatory medication,
Sinus Aspergillosis J. Cranio-Max.-Fac. Surg. 18 (1990) 35

fistula closed spontaneously. On admission, left maxillary presence of dense particles in maxillary sinus. Medial si-
sinus painful, diminished radiolucency but absence of bone nus: and orbital walls destroyed (Fig. 3). Biopsies negative.
destruction. Caldwell-Luc sinus debridement performed Through a lateral rhinotomy, all left paranasal sinuses
and pathological confirmation of diagnosis of Aspergillo- were cleared. Several biopsies and cultures taken from and
ma on mastic-like material. N o relapse in 9 years. around mastic-like mass. The pathological diagnosis ex-
Ca~e 2: 31-year-old caucasian woman, oro-antral fistula cluded malignancy and indicated S. A. Aspergillus found in
after extraction of the 16 three months earlier. Several an- cultures of the specimen. No relapse in 4 years.
tibiotic courses and sinus-washes with Soframicin | with- Case 9: 60-year-old caucasian woman with long-standing
out success: Pain and x-ray findings (opacification, no diabetes, not kept under proper medical control. Recur-
bone destruction) indicated right maxillary sinusitis. Cald- rent frontal headaches (especially in tile supine position),
well-Luc operation performed and pathological examina- and recently vomiting, hiccup and loss of consciousness
tion revealed Aspergillosis. No relapse in 6 years. (probably due to hypopotassaemia). N o evidence of intra-
Case 3: 30-year-old african male, a Behqet-patient, under cranial hypertension. CF-scan showed opacity of the left
medication (colchicine, aspirin, ticlide) for 10 months, sphenoid- and ethmoid sinuses on both sides, with clear
showing chronic uv~itis in both eyes and presenting striae, running through tile cribriform plate into the dura
asymptomatic left maxillary sinusitis. Dense bodies (diam. and frontal lobe. These appearances suggest Aspergillus-
3 mm) on x-ray in opaque sinus and clear perforation of filaments, as an alternative diagnosis to malignancy. Later-
root-canal filling of tooth 26 with eugenate (zinc-oxide- al rhinotomy performed on either side, and spheno-eth-
eugenol) overflow into the maxillary sinus. Apicectomy moidectomy performed up to dura. Biopsy specimens
w i t h surgical debridement of the sinus (Caldwell-Luc). Di- showed typical striae of Aspergillus. Initially, patient did
agnosis confirmed by pathological examination. No re- well under broad sprectrum antibiotics (Penicillin + Me-
lapse in 4 years. tronidazole). After control of diabetes, patient transferred
Case 4: 35-year-old caucasian woman, badly performed to another hospital. Patient died 6 months later of a cereb-
root-canal treatments on 25 and 27. Treatment with i.v. rovascular accident, CT images showing intracranial
Penicillin + Metronidazole tw9 months before admission spread of Aspergilloma (Fig. 6). N o autopsy was per-
because of extensive cellulitis of left cheel<. On x-ray, me- formed.
tal-like dense bodies (diam. 4 mm) present in left maxillary Case 10: 56-year-old obese caucasian male suffering from
sinus. Caldwell-Luc performed, no relapse in 1 year. Diag- rhinorrhoea, nasal obstruction (predominantly on right
nosis confirmed by pathological examination. side), and sinus fullness for one month. Moderate septal
Case 5: 35-year-old caucasian woman with chronic left deviation to the right, due to old nasal trauma. No history
maxillary sinusitis, present for 12 months and left untrea- of sinus problems, no medication, surgical history limited
ted. Root-canal treatment of tooth 26, 18 months previ- to abdominal operations. Diminished translucency of right
ously. Metal-like dense bodies in sinus on x-ray. Caldwell- maxillary sinus and presence of metal-dense foreign body
Luc operation performed and no relapse in 7 years. Patho- (diam. 6 mm) in centre of opacity. Root of tooth 13 ex-
logical confirmation of diagnosis of S. A. tended into right maxillary sinus showing a poorly per-
Case 6: 30-year-old caucasian woman, posterior nasal formed rootcanal filling. Left maxillary sinus also showed
drip. N o evidence of dental factor. Sinns puncture per- a local opacity, indicating a sinus polyp. Bilateral Cald-
formed three months earlier (culture negative). Antibiotics well-Luc, extraction of tooth 13 and correction of the de-
and local steroid therapy gave temporary improvement. viated septum. Mastic-like material found only in right
Standard x-rays and CT-scan now show presence of metal- maxillary sinus, revealing Aspergillosis on pathological ex-
dense body within confines of opaque left maxillary sinus. amination. Left maxillary sinus polyp negative for fungi.
Caldwell-Luc performed and no relapse after 2 years. As- Culture on Sabouraud's agar of all curretted materials re-
pergillosis diagnosed on pathological examination. Rou- mained negative. No relapse over 5 months.
tine culture remained negative.
Case 7: 36-year-old woman with posterior nasal drip. Si- Results
nus puncture 2 months previously (culture negative). Anti-
biotic and anti-histamine therapy without result. Radio- In each centre we have found five cases of Sinus Aspergil-
logical evidence of eugenate-paste floating free in the left losis: Cases 1-5 in the Paris unit and cases 6 - 1 0 in the
maxillary sinus, after root-canal treatment of tooth 26. Leuven unit (Table 2). In this series:
Inflamation extended into opaque ethmoid sinus (strictly - 80 % of the patients were in their fourth decade, 20 %
left unilateral) and perforated medial wall of left maxillary older "
sinus. Caldwell-Luc procedure was combined with eth- - 70 % were female
moid sinus curettage followed by one month of Nizoral| - 70% were non-invasive forms according to Hora's
(1 tablet daily). Diagnosis (and presence of eugenate) cou- (1965) classification (cases 7, 8, 9)
firmed by pathological examination. No relapse in 2 - in 70% there was evidence of contamination of the
years. maxillary sinus, (71% by eugenate and 29 % via an oro-
Case 8" 57-year-old caucasian man with strictly unilateral antral fistula), vs. 20% in which there were circum-
pansinusitis, invasion of left medial wall of maxillary sinus stances indicating a higher probability of spore inhala-
and orbit. Chronic pain in left maxillary sinus (onset 4 tion (case 8 - tourism, case 9 - farmer's wife). 10%
months earlier) spread to retrobulbar area. Chronic poste- could not be accounted for.
r i o t nasal drip. Poor periodontal condition indicating pos- Treatment of our 10 cases comprised removal of all the
sible dental origin. Recent holiday in India. Several courses pathological sinus mucosa. In most cases tile currettage
of broad-spectrum antibiotic therapy. Radiographs and was done through a classic Caldwell-Luc approach. As a
CF-scan showing opacity of all sinuses on the left with routine, the currettage was limited to the pathological area
36 J. Cranio-Max.-Fac. Burg. 18 (I990) Christophe De Foer et al.

of the sinus mucosa. Only cases 7, 8 and 9 required more


extensive treatment.

Discussion
Aspergillus is a fungus of the species of Ascomycetes,
which is found in different kinds of soil. Within the group
of fungi, Aspergillus is by far the commonest to produce
infection of the nose and paranasal sinuses (Savetsky and
Waltner, 1978). Three different subtypes are regarded as
possible parasites or saprophytes of the nose: A. Flavus, A.
Fumigatus and (rarely) A. Niger (Axelsson et al., 1978).
More than 90 % of the so-called "Aspergilloses" are pul-
monary infections. Besides, the respiratory tract is the
main "porte d'entr~e" for generalized A. infections, which
secondarily spread to other organs (Schrijver et al., 1983).
In contrast to this "metastatic" infection, the sinus is al-
most exclusively infected primarily; i.e. by direct inhala-
tion of the spores. Milosef and Mahgoub (1969) reported
on the largest series observed to date. Their 69 patients
were recruited in the tropical Sudan, where the disease is
endemic because of the high density of Asp. spores in the
dust and the frequent sand-storms. Other authors have
confirmed the favourable influence of a warm and moist
climate on the development of S.A (Hora, 1965; Warder et
al., 1976; McGuirt and Harrill, 1979; McGill et al.,
1980). The spores of A. flavus are found in the nasal se-
cretion of most of the Sudanese population.,Many Asp. in-
fections here develop in a spectacular way probably as a
result of the relatively late request for medical care (Milo-
sef and Mahgoub, 1969; Mahgoub," 1971; Veress et al.,
1973). However difficult it seems to exclude contact with
"atmospheres at risk" of spore-inhalation (increasing air-
traffic to Africa, as was seen in a case by Lowe and Brad-
ley in 1986; or tourism in India, as seen in our case no. 8),
Fig,4 Light micrograph of the Aspergillus-colony (P.A.S.-cofour- it is likely that another pathogenic mechanism is involved
ing, magnific x 15), showing the typical septate hyphae and the As-
pergillus heads, directed toward the periphery of the fungal mass
in the development of S.A. in our European conditions.
(case 9).

Table2 A survey of the 10 presented case reports

sex age site CT/x-ray assoc, path. factor peroperative finding culture

case 1 f 31 L. Max O dental implant mastic-like material n.m.


case 2 f 31 R. Max O oroantral fistula n.s. n.m.
case 3 m 30 L. Max O, D.P. Beh~et/eugenate mastic-like material n.m.
case 4 f 35 L. Max O, D.P. rootcanal perfor? n.s. n. m
case 5 f 35 L. Max O, D.P. ro~tcanal perfor? n.S. n.M.
case 6 f 30 L. Max O, D.P. -- n.s. neg.
case 7 f 36 L.M., E. S O, F. B., B.D. rootcanal perfor eugenate neg.
case 8 m 57 L.M., E., S, Orbit O, B.D. periodontitis/India mastic-like material Asperg.
case 9 f 60 Bilat E,, S. Brain O.B. D diabetes/farming mastic-like material n.m.
case 10 m 57 R. Max O, D.P. -- n.s. neg.
case 11 f 56 R. Max O, D.P. -- "~ mastic-like material n.m.

R. M(ax) = Right Maxillary Sinus


L.M, E, S, Orbit = Left Maxillary, Ethmoid and Sphenoid sinus and Orbital invasion
O = opacification of sinus on CT-scan or standard x-ray
D.P. = metaldense particles
'~ F.B. = foreign body (eugenate fragment)
B.D. = bone destruction
n.m. = not ment~0ned
n.s. = not specified
Sinus Aspergillosis J. Cranio-Max.-Fac. Surg. 18 (I990) 37

Legent et at. (1980) observed several cases of S.A. after


perforation of the maxillary sinus during endodontic
treatment in the maxillary dentition. In 50 % of 20 maxil-
lary Asp. sinusitises, eugenate paste was found floating
free in the maxillary sinus. This might indicate a probable
cause of chronic Asp. sinusitits. Moreover, in all 20 cases,
some factor in the maxillary dentition was found to be a
possible cause of the infection, the majority of patients be-
ing in good'general condition (Legent et al., 1980, 1982,
1984).
Legent comments on his hypothesis as follows:
-throughout the literature, the unilateral maxillary
localisation of S.A. is by far the most frequent one.
Though sometimes it is part of a pansinusitis, isolated
ethmoidal-, sphenoidal-, or frontal- sinusitis is rare (only
5 %, 13 % and 2 % respectively if the Sudanese cases are
not taken into account). (Schrijver et al., 1983, Glass,
1984).
- The combination of the immunosuppressive effect of
corticosteroids, when added to eugenate obturator-
pastes would especially favour the development of a
S.A.
Legent et al. (1982) do not exclude the possible climatic
factor. According to the authors, certain regions in France Fig.5 Sinus Aspergillosis, aetio-pathogenesis, possible develop-
(Bordeaux, Nantes) would have more cases to report than ment.
others. First pathway: direct spore-inhalation via nose
It is our opinion that both the "aerogenic" and the "den- - traditionally observed in (sub)tropical climats
togenic pathways can explain port of the pathogenesis of - explains (the rare) ethmoid and sphenoid S.A.'s.
S.A. Depending on clinical and radiological observations - impossible to exclude in any case, since spores omnipresent
(e. g. panoramic and tomographic images of the maxillary Second pathway: (iatrogenic) maxillary sinus contamination during
dentition), as well as the circumstances in which the pa- manipulations in maxillary dentition (implants, fistula, rootcanals)
tient lives (e. g. urban vs. rural area, profession, air-travel - probably more important in industrialised, urban area
to other climates, hygiene), the clinician may come to a - explains the predominantly unilateral maxillary sinus Iocalisation
correct attribution of the origin of the infection. In our 10 - does not explain why the majority of (iatrogenic) sinus perfora-
cases, either pathway seems to contribute to tile explana- tions do not develop S.A.-additional hygienic factor?
tion of the totality of the series. Possible invasion of vital structures
1) Orbit
In seven cases there was evidence of a "dental" factor (5 2) cribriform plate, brain
endodontic sinus perforations (case 3, 4, 5, 7, 10), 2 oro-
sinusal fistula (case 1, 2)).
Clinically, S.A. should be thought of in case of:
a) every sinusitis, that does not respond to antibiotics and
irrigation;
b) blood or greenish, grayish membranes (mastic-like) on
antral wash-outs; Fig.6 CT-scan
c) every strictly unilateral sinusitis; image showing in-
d) destruction of the bony sinus walls. tra-cranial spread
Radiologically, S. A. presents as an unilateral loss of trans- of the Aspergillo-
ma, after direct de-
lucency of the affected sinus. Eventually, metal-dense par-
struction of sphe-
ticles can be observed within tile confines of this opacity. noid bone (case 9).
They would be due to high calciumphosphate in tile in-
tracellular milieu of the necrotising Asp. cells (Kopp,
1985; Stammberger, 1985). We have observed such dense
particles on different radiographs (see figures 1 and 2) of
seven of our cases (3 to 8 and 10). However, all these par-
ticles were seen in the maxillary sinus, and in several cases,
they might well have been foreign bodies (e. g. eugenate in
case 3, 4, 5, 7 and 10).
Nevertheless, in cases 6 and 8, metallic-dense particles
were observed, while there was no context of endodontic
treatment in the neighbouring maxillary dentition. Zin-
reich et al. (1988) diagnosed 22 of 25 (76%) cases of fun-
gal sinusitis on CT images. They stated that the findings
on Magnetic Resonance Imaging would appear even more
characteristic of fungal sinusitis. They typical increased at-
38 J. Cranio-Max.-Fac. Surg. 18 (1990) Cbristopbe De Foer et al.

tenuation on a CT and the hypointense signal on an MRI (Katzenstehl, 1983). In the invasive cases, the surrounding
would be due to high concentrations of Calcium, Iron and tissues show signs of an immunological reaction, such as
Manganese in the mycetoma. fibrinoid changes of the blood vessels and extravasation of
Most often, the evolution of S.A. is benign, and resembles lymphocytes; pseudotuberculae similar to those seen in
any chronic bacterial sinusitis. The most important distinc- other conditions of delayed hypersensitivity. The sinus is
tive characteristic however, is its potential to become inva- filled with a mucoid mass, in which eosinophylic cells, leu-
sive at any stage. When confronted with a pseudo-tumou- cocytes and Charcot-Leyden crystals are scattered through-
ral process in the maxillary sinus (cholesteatomas and out the hyphae (Veress et al., 1973).
mugoc6eles are predominantly found in fronto-ethmoidal The only effective treatment of S.A. is total debridement
sinuses), one should always consider the possibility of an of the affected sinus(es). Depending on the clinical and ra-
invasive S. A-(Juvanon et al., 1987). diological findings, access will be gained through a trans-
lnvasive S.A. is a rare disease, but can be seen in patients oral antrostomy or through a lateral rhinotomy. Sinus-
in otherwise perfect physical condition. Immunodepres- wash-outs and/or conservative antibiotic-therapy are never
sion enhances the chances of possible invasiveness (Warder effective. Any chronic maxillary sinusitis, showing one or
et al., 1976; Vanderstock et al., 1981). However, invasion more of the characteristics mentioned above should be
of the brain and orbits in healthy patients has been report- treated by surgical debridement without delay. Even
ed several times (Young et al., 1970; Jahrsdoerfer et al., though many iatrogenic (e. g. implantological, endodontic)
1979; Bahadur et al., 1983; Yumoto et al., 1985; Lowe sinus perforations remain trouble free, there is evidence
and Bradley, 1986). that in case of oro-sinusal fistula or spilling of eugenate-
Also in our series, two of our three invasive cases were in paste into the sinus, a conservative approach (antibiotics
perfect physical condition. These cases (case 7 and 8) sus- and corticosteroids) will enhance the (possibly invasive)
tain the conclusions that invasive S.A. may occur in heal- proliferation of the fungal mass (hvamoto et al., 1972;
thy subjects. In the third case (case 9) there was evidence of Bambule and Grigoriu, 1982; Bassiouny et al., 1982). As
impaired defences (i. e. diabetes). In this case the outcome illustrated in our cases 7 and 8, surgery is also effective in
of the intracerebral spread of the infection was, despite in- tile invasive forms. Radiation therapy is obsolete.
tensive treatment, fatal to the patient. Some discussion exists on the additional use of Amphoteri-
The invasion of surrounding tissues is sometimes charac- cin B after surgery. However much its in vitro effective-
terised by pain (invasion of overlying skin) but can also ness has been proved, the clinical use of Amphotericin B is
present as a painless, expanding, firm mass. Invasion of the less evident.
orbit is characterised by impaired vision and/or eye mobili- Stevens (1981)suggested the use o f hyperbaric oxygen in
ty, exophthalmia and temporo-occipital headaches. In the S.A., but to our knowledge, clinicfil reports of its use in
case of ethmoidal invasion, the process will expand (as S.A. have not yet been published. In Mucormycosis, ad-
does a carcinoma) via the path of least resistance, that is junctive hyperbaric oxygen therapy has already been
the cribriform plate. Intra-cranial extension (as in case 9), proved effective after surgical debridement. The rationale
will very often have a fatal outcome. The differential diag- for its use is that this treatment modality would increase
nosis between S.A. and a carcinoma on radiographic find- tissue oxygen concentration and reduce acidosis, even in
ings is a difficult, if not impossible task. Sometimes the poorly perfused tissues.
circumstances can be very misleading, as reported by Bris- Farmer (1985) stated that, in view of the results already
seau (1986): he reported a case of ethmoid S. A. in a wood- obtained by others, adjunctive hyperbaric oxygen should
worker, a profession known to be predisposed to adeno- be used in rhinocerebral Mucormycosis.
carcinoma in the ethmoid sinus. On the other hand, Asper- Indeed, in vitro studies by several investigators have shown
gillosis can coexist with a carcinoma in the sinus. In such a that oxygen in sufficient concentrations is fungistatic for
case one wonders which was the primary pathology (Tano- Mucor and Aspergillus. We suggest that hyperbaric oxy-
ka, 1985)? Another differential diagnosis to be considered gen be used in invasive cases of S.A., when there is evi-
is Mucormycosis. This presents as an obstructive mass, in- dence of extensive necrosis, and only as an adjunctive ther-
volving the sinus in a way similar to Aspergillosis. Peroper- apy to surgery. It seems obvious that, since Aspergillus
atively, a comparable "mastic-like" material is found and it lacks the predilection for invasion of blood vessels (charac-
can become as invasive. teristic of Mucormycosis), resulting in thrombosis and in-
The diagnosis of Aspergillic infection is based on the mic- farction of the tissues, the usefulness of oxygen is doubtful
roscopic findings. Aspergillus has narrow (41am) septate in S.A. In most cases, aeration of the sinus is a necessity
hyphae (Fig. 4), in contrast to Mucormycosis which has. but t'he use of a hyperbaric oxygen chamber is cumber-
broader (6 to 50 [tm) rarely septate hyphae of uneven dia- some and superfluous. The prognosis of S.A. fully de-
meter. Also, destruction of blood vessels (with eventually pends upon the early diagnosis and accurate surgery.
haematogenous spread) is typical of Mucormycosis, since Unfortunately, serological screening is, in contrast to pul-
Aspergillus does not invade blood vessels. monary Aspergillosis, completely unreliable in S.A.
A positive culture on Sabouraud's agar does not provide To some extend, the subtype of Aspergdlus 0. e. the more
exact proof of the Asp. infection since spores are omni- virulent A. Flavus) and the site of the expanding mass (i. e.
present in the atmosphere. However, culturing "fresh" proximity of cribriform plate or optical nerve-canal) are
curretted material is o f additional value in identifying the" decisive for the prognosis of the disease. Intracranial inva-
subtypes. sion most often has a fatal outcome. (Gupta et al., 1973;
The microscopic image of an Aspergilloma shows (after Miglets et al., 1978).
staining with P.A.S. or-Gomori-Grocott) the typical
branched hyphae of the filaments, with the heads of the
Aspergilli, confined to the periphery of the fungal mass
Sinus Aspergillosis J. Cranio-Max.-Fac. Surg. 18 (1990) 39

Conclusions lwamoto, H., M. Katsura, T. Fujimaki: Mycosis of the maxillary si-


nuses. Laryngosc. 82 (1972) 903-909
I n . c o n t r a s t to Mucormycosis, which is often discussed in Jabrsdoerfer, IL, V. Ejercito, M. Johns, R. Cantrell, Sydnor: Aspergil-
the oto-rhinolaryngological literature, Sinus Aspergillosis losis of the nose and paranasal sinuses. Am. J. Otolaryngol. 1
is a rarely r e p o r t e d disease. One wonders if M u c o r is really (1979) 6-14
a m o r e frequent infection than Aspergillus. It is o u r guess Juvanon, J. M., M. Feuilhade de Chauvin, I. Souchal Delacour, C.
that most o f the cases of Sinus Aspergillosis seen in o u r Brun Buisson, tL Peynegre: Aspergillose naso-sinusienne grave
countries are first seen by oral surgeons, since the basis o f chez l'immunod~prim6, ~ propos de trois cas. Ann. Otolaryngol.
104 (1987) 369-374
the p r o b l e m often resides in the maxillary dentition. This
Katzenstein, A.: Pathologic findings in allergic Asp. sinusitis. Am. J.
is in corfirast to the large series of " a i r b o r n " forms previ- Surg. Pathol. 7 (1983) 439-443
ously described in tropical countries. The majority o f these Kopp, W.: Metal dense structures in the paranasal sinuses. Rofo
S.A.'s was caused by more virulent species (A. Flavus) in a 142/3 (1985) 288-290
different p o p u l a t i o n and climate. In our series, the majori- Kopp, W.: Asp. of the paranasal sinuses. Radiol. 156/3 (1985)
ty o f cases were relatively "tranquil" forms with a dental 715-716
history. Possibly, they represent the "tip o f the iceberg" o f Legent, F., C. Beauvillahz, J. Mercier, B. Hofinann, M. Wesolucb:
a multitude o f Aspergillic sinusitises that are treated as an Rapports entre l'Asp, sinusienne, les rhinolithes primitifs et la pa-
o r d i n a r y sinusitis. thologie dentaire. Ann. d'otolaryngol. 99 (1982) 541-545
Legent, F., J. J. Boutet, G. Hofi~tann, H. Massot, J. Galiba: Aspergil-
T w o o f o u r three invasive cases seemed to show an a i r b o r n
loses sinusiennes. Rev. pratic. 34 (1984) 2105-2109
c o n t e x t rather than a dental one. There might be a differ- Legent F., Desnos, J., C. Beauvillain, G. Trichereau, J. M. Julien M.
ence in virulence o f the species that are inoculated via the Bout, C. Vermeil: Aspergilloses sinusienes. J. Franc. d'Oto-Rhyno-
" a i r b o r n " instead of the "dental" p a t h w a y . It w o u l d be in- Laryngol. 29 (1980) 39-49
teresting to investigate and culture maxillary sinusitises Lowe, J., J. Bradley: Cerebral and orbital Asp. infection due to inva-
m o r e often, especially if metal-dense particles are seen in sive Aspergillosis of ethmoid sinus. J.Clin. Pathol. 39 (1986)
the sinus on a radiograph. A high index o f suspicion of the 774-778
diagnosis will p r o b a b l y reveal m o r e S.A. infections in the McGill, T. J., G. Shnpson, G. B. Healy: Fulminant Aspergillosis of
nose and paranasal sinuses; a new clinical entity. Laryngosc. 90
future. "
(1980) 748-754
McGuirt, W., J. A. Harill: Paranasal sinus Aspergillosis. Laryngosc.
Acknowledgements 89 (1979) 1563-1568
The authors wish to express their sincere thanks to Dr. G. van Parijs Mabgoub, E.: Mycological and serological studies on Asp. Flavus
and Prof. van Damme (Dept. of Pathology - U.Z. Leuven), for their isolated from paranasal Asp. in the Sudan. J. Trop. Med. Hyg. 74
excellent micrographic photographs and advice on the pathology. (1971) 162-165
Especially, they thank Dr. A. Cbarbit for his well-kept files on the Miglets, W., W. Saumters, L. Ayers: Aspergillosis of the sphenoid si-
five Parisian cases which he had included in his thesis on the subject. nus. Arch. Otolaryngol. 104 (1978) 47-45
Milosef, B., F_.Mahgoub: Primary Asperg. of paranasal sinuses in the
Sudan. Brit. J. Surg. 56 (1969) 132-13
Ragot, J. P., P. Noivet, S. Imbert: Aspergillose du sinus maxillaire,
les pStes d'obturation canalaire sont-elles responsables? A.E.O.S.,
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