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Defoer 1990
Defoer 1990
Defoer 1990
18 (I990) 33
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).
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.
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).
sex age site CT/x-ray assoc, path. factor peroperative finding culture
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
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B-2060 Antwerp in der Scbonau
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