Download What`s in a fungus ball? Report of a case with

ORIGINAL
ARTICLE
SENOCAK,
KAUR
What’s in a fungus ball? Report of a
case with submucosal invasion and
tissue eosinophilia
Dogan Senocak, MD; Ahmet Kaur, MD
Abstract
Fungus balls are tangled mats of hyphae that are often
found in the maxillary sinus. In approximately half of affected patients, radiologic evaluation will reveal areas of
hyperdensity within soft-tissue masses. Histopathologic
examination will reveal no invasion of the mucosa and
no granulomatous reactions. Surgical removal is sufficient because fungus balls are not known to recur. We
describe an interesting case of a sinonasal fungus ball that
resembled dental filling material on radiologic imaging
because of its extraordinary radiopacity. Histopathologic
examination detected eosinophilic infiltration, hyphae in
the submucosal tissues, and tissue necrosis.
Introduction
The introduction of the concept of allergic fungal rhinosinusitis and the publication of recent studies showing a
high incidence of fungal matter in histologic specimens
taken from patients with chronic rhinosinusitis have led
to a good deal of debate lately.1
Clinically, a patient’s immune status determines the
degree of fungal infection of the paranasal sinuses.2
Patients with invasive fungal rhinosinusitis are usually
immunocompromised, and therefore they are likely to
experience potentially fatal complications. On the other
hand, allergic fungal sinusitis is thought to be the result
of an atopic reaction to the causative fungus, and most
patients with fungus balls are immunocompetent. Growth
of saprophytic fungi has also been seen on the mucosa and
crusts in the sinonasal cavity of immunocompetent patients
following sinus surgery.
Fungus balls are tangled mats of hyphae that are frequently found in one sinus only, most often the maxillary
sinus.3 In the rhinologic literature, they are often referred
Dr. Senocak is with the Department of Otolaryngology–Head and Neck
Surgery, Cerrahpasa School of Medicine, Istanbul University. Dr.
Kaur is a pathologist in private practice in Istanbul.
Reprint requests: Dogan Senocak, MD, BM Pasa Sok. 1/7, Aslanli Apt.,
Etiler, Istanbul, Turkey. Phone: 90-532-321-6402; fax: 90-212-2631388; e-mail: [email protected]
696
to as mycetomas. In approximately half of affected patients,
radiologic evaluation will reveal areas of hyperdensity
within soft-tissue masses. These areas are believed to
represent dense hyphae or metallic depositions. Histopathologic examination will reveal no invasion of the
mucosa and no granulomatous reactions. There has been
1 report of a fungus ball that became invasive after the
patient became immunosuppressed as a result of a kidney
transplant.4 Surgical removal is sufficient because fungus
balls are not known to recur.
In this article, we describe an interesting case of a sinonasal fungus ball that resembled dental filling material on
radiologic imaging because of its extraordinary radiopacity.
Histopathologic examination detected eosinophilic infiltration, hyphae in the submucosal tissues, and tissue necrosis.
These findings perhaps represented the initial phase of an
invasive progression.
Case report
A dentist had treated the upper left canines of a 64-year-old
woman. Shortly thereafter, the woman began to experience left facial pain. The dentist obtained a panoramic
x-ray, which demonstrated a bright lesion in the left maxillary sinus (figure 1). Believing that this lesion probably
represented dental paste in the sinus cavity, the dentist
referred the patient to our facility for sinus exploration.
The referral was made 3 months following the original
dental procedure.
Findings on physical examination were normal except
for the presence of left infraorbital pain on pressure. Computed tomography (CT) of the paranasal sinuses revealed
the presence of a large concha bullosa on the left, mucosal
thickening on the left, and a very bright 3 × 5-mm mass
in the left maxillary sinus (figure 2). The brightness of the
lesion was almost identical to that of the patient’s dental
fillings, suggesting that they might very well have been
made of the same material.
We advised the patient that because the lesion was large
enough to eventually obstruct the sinus ostium, surgery
was justified, and the patient consented. We approached the
left maxillary sinus endoscopically through the enlarged
ENT-Ear, Nose & Throat Journal ■ October 2004
WHAT’S IN A FUNGUS BALL? REPORT OF A CASE WITH SUBMUCOSAL INVASION AND TISSUE EOSINOPHILIA
Discussion
There are a few interesting aspects of this rather simple and
straightforward case that make its publication worthwhile.
The brightness of the maxillary sinus lesion and the patient’s
recent history of dental work suggested that the mass might
very well have been a foreign body. If that had been the
case, would surgical intervention have been necessary to
remove it? We based our surgical indications on the size
of the lesion (which was large enough to obstruct the sinus
ostium) and the mucosal reaction around it (a potential
cause of chronic sinusitis). If the patient had been unfit for
surgery or if she had refused it, we probably would have
opted for a wait-and-see approach. However, because the
patient was in the initial phase of an invasive process, this
would have proved to be the wrong decision.
The literature contains several reports on the CT diagnosis
of fungal sinusitis5-7 and the effects of zinc oxide-euginol
paste.8-11 Some authors strongly believe that CT studies—especially CT densitometry—are important tools in making
the differential diagnosis of fungal sinusitis and foreign
bodies of dental origin in the sinus; others claim that there
are no valid predictive criteria for CT diagnosis. However,
none of these authors has commented on the necessity of
CT to validate surgery. An effective method of differentiating fungal growth from a foreign body preoperatively
would be very useful, especially in cases of unsuspected
submucosal invasion, such as occurred in our patient.
Articles on the effects of zinc oxide-euginol paste in
the sinus are more interesting from a rhinologist’s point
of view because some authors suggest that it is a cause of
fungal growth. This notion is not commonly recognized
by otolaryngologists, and perhaps it deserves to be better researched by sinus surgeons. Although some authors
believe that zinc is both the cause of aspergillar growth in
the sinus and the reason for radiopaque foci, one controlled
study on the effects of zinc oxide-euginol paste failed to
show that it acts as a growth factor for Aspergillus.11 In
our literature review, we did not find any studies on what
constitutes the radiopacity of fungal masses in the sinuses.
Sulfur granules or calcifications have been suggested as
possible causes.
The mucosa surrounding the fungus ball in our patient
was sent for histopathologic evaluation. Areas of fungal
growth in the mucosa could be seen macroscopically. Microscopic examination revealed that what had appeared
to be submucosal invasion was mostly hyphae and spores
covered by necrotic epithelium and eosinophils; there were
also areas of submucosal hyphae extension and spores.
Figure 2. Coronal CT of the paranasal sinuses shows the hypertrophic mucosa and the very bright lesion in the left maxillary sinus. Note that the lesion is just as bright as the patient’s
dental fillings.
Figure 3. Macroscopic image of the polypoid mass shows the
pigmented area.
Figure 1. Panoramic dental x-ray clearly shows the bright lesion
in the left maxillary sinus.
natural ostium. The lower third of the maxillary sinus was
filled with polypoid mucosa that peeled off easily from the
underlying mucoperiosteum. In the center of the polypoid
tissue was a brown, crusty lesion that was macroscopically identified as an Aspergillus mycetoma. The polypoid tissue also featured an area of pigmentation (figure
3). Histopathologic examination of the tissues revealed
extension of hyphae into the submucosa and the presence
of submucosal necrosis, findings that are consistent with
aspergillosis (figure 4). A complete blood analysis failed
to reveal any immune system failure.
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SENOCAK, KAUR
HEAD AND NECK CLINIC
Continued from page 684
Figure 4. Histopathologic study of the specimen reveals eosinophilic infiltration (single arrow), branching septate aspergillar
hyphae (double arrows), submucosal necrosis (triple arrows),
and fruiting bodies of an Aspergillus colony (quadruple arrows)
(H&E, original magnification × 400).
This case represents a rather unusual expression of
all three possible types of fungal sinusitis in 1 patient
(a fungus ball extending submucosally with a strong eosinophilic reaction around it). To our knowledge, no such
case has been published before. The reason might be that
most fungus balls are easily diagnosed macroscopically,
so histopathologic examination is not usually performed.
Whatever the case may be, histopathologic examination
of every specimen removed from the sinuses appears to
be necessary if we are to understand the involvement of
fungal pathogens in rhinosinusitis. This single case, with
all its complexities, provides a good lesson in fungal rhinosinusitis, and it illustrates how much more we have to
learn about it.
References
1. Ponikau JU, Sherris DA, Kern EB, et al. The diagnosis and incidence
of allergic fungal sinusitis. Mayo Clin Proc 1999;74:877-84.
2. Ferguson BJ. Definitions of fungal rhinosinusitis. Otolaryngol Clin
North Am 2000;33:227-35.
3. Ferguson BJ. Fungus balls of the paranasal sinuses. Otolaryngol Clin
North Am 2000;33:389-98.
4. Gungor A, Adusumilli V, Corey JP. Fungal sinusitis: Progression of
disease in immunosuppression—a case report. Ear Nose Throat J
1998;77:207-10, 215.
5. Lenglinger FX, Krennmair G, Muller-Schelken H, Artmann W. Radiodense concretions in maxillary sinus aspergillosis: Pathogenesis
and the role of CT densitometry. Eur Radiol 1996;6:375-9.
6. Dhong HJ, Jung JY, Park JH. Diagnostic accuracy in sinus fungus balls:
CT scan and operative findings. Am J Rhinol 2000;14:227-31.
7. Lund VJ, Lloyd G, Savy L, Howard D. Fungal rhinosinusitis. J Laryngol
Otol 2000;114:76-80.
8. De Foer C, Fossion E, Vaillant JM. Sinus aspergillosis. J Craniomaxillofac Surg 1990;18:33-40.
9. Zimmerli P, Hardt N, Altermatt HJ. [Maxillary sinus operation. Aspergillosis of the maxillary sinus caused by dental root filling materials].
Schweiz Monatsschr Zahnmed 1988;98:527-30.
10. Legent F, Billet J, Beauvillain C, et al. The role of dental canal fillings
in the development of Aspergillus sinusitis. A report of 85 cases. Arch
Otorhinolaryngol 1989;246:318-20.
11. Odell E, Pertl C. Zinc as a growth factor for Aspergillus sp. and the
antifungal effects of root canal sealants. Oral Surg Oral Med Oral Pathol
Oral Radiol Endod 1995;79:82-7.
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Figure 3. Photograph shows the immediate postoperative
result.
gradually freed from the other perioral muscles (muscles
of facial expression) in order to maximize advancement of
the flap. Care is taken to locate and preserve the superior
and inferior labial arteries, as well as the branches of the
facial nerve that enter the orbicularis oris peripherally and
deep to the muscle. Once mobilization is complete, a meticulous tension-free, three-layered closure is performed,
and care is taken to reapproximate the vermilion (figure
3). Complete and immediate function may be restored
because the neurovascular pedicle is preserved.
Although the Karapandzic flap is no more beneficial
than other flaps in terms of microstomia, it does offer
several advantages:
• It allows surgeons to fill a lip defect with tissue that is
similar to that of a nascent lip.
• Circumoral scars may be cosmetically acceptable
because they are located in the nasolabial and mental
creases. Cosmetic appeal may be limited in patients with
large defects. Crescentic perialar incisions may be used to
decrease the tissue irregularities that occur at the nose.
• The Karapandzic flap results in better oral competence
because it preserves motor and sensory innervation. The
labial arteries and the facial nerve branches along the periphery of the orbicularis oris are skeletonized and preserved,
which allows for the maintenance of their function.
• The flap can be created in a single stage.
Suggested reading
Karapandzic M. Reconstruction of lip defects by local arterial flaps. Br
J Plast Surg 1974;27:93-7.
Panje WR. Lip reconstruction. Otolaryngol Clin North Am 1982;15:
169-78.
Polly AD, Tan EP. Lower lip reconstruction. Br J Plast Surg 1981;34:
83-6.A
ENT-Ear, Nose & Throat Journal ■ October 2004