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FiedlerLS, WunschA. BMJ Case Rep 2021;14:e241487. doi:10.1136/bcr-2020-241487
Ameloblastoma of the maxillary sinus: conservative
surgical management considering high recurrence
riskpotential
Lukas S Fiedler , Annette Wunsch
Case report
To cite: FiedlerLS,
WunschA. BMJ Case
Rep 2021;14:e241487.
doi:10.1136/bcr-2020-
241487
Otorhinolaryngology and Head
and Neck Surgery, Klinikum
Mutterhaus der Borromäerinnen
gGmbH, Trier, Deutschland,
Germany
Correspondence to
Dr Lukas S Fiedler;
l. fiedler@ gmx. at
Accepted 28 April 2021
© BMJ Publishing Group
Limited 2021. No commercial
re- use. See rights and
permissions. Published by BMJ.
SUMMARY
Ameloblastoma (AM) in the maxillary sinus is rare. This
benign entity shows locally invasive, destructive and
aggressive behaviour and a high rate of recurrence.
Therefore, the course of treatment is radical resection.
We report the case of a 38- year- old man presenting with
signs of recurrent sinusitis in the Ear, Nose and Throat
Department. Transnasal flexible endoscopy revealed
a cystic mass in the right inferior and middle nasal
passage. CT scan showed an obliterated right maxillary
sinus with a ballooning effect and pressure atrophy of
the lateral sinus wall, without possible differentiation
of the middle and low nasal turbinate. The patient
was treated with transnasal functional sinus surgery;
pathology stated AM. AM in the maxillary sinus is rare,
locally destructive and therefore as a gold standard is
resected radically to prevent recurrence. We demonstrate
a conservative approach; explicitly, we combined a
transvestibular and functional endoscopic sinus surgery
resection of the AM to maintain function and reduce
the possibility of postoperative impairments. Whether
the strategy of treatment for AM is conservative, it
nonetheless can result in a recurrence- free status.
Nevertheless, inclusion into an oncological follow- up-
programme with regularly performed MRI and CT is
recommended.
BACKGROUND
Ameloblastoma (AM) is a very rare, benign, locally
invasive tumour that can affect the sinuses and
shows a high recurrence rate. Therefore, the gold
standard therapy is radical resection.
CASE PRESENTATION
A 38- year- old man presented to an Ear, Nose and
Throat Department in a community care hospital
due to symptoms of recurrent sinusitis, which
subjectively started after an infection of the upper
airway. Since then, he suffered from nasal obstruc-
tion. Conservative treatment with a corticoid spray
for 1 month brought no improvement. Further-
more, medical anamnesis was clear, no relevant
medical event was recorded and laboratory results
showed no pathology.
INVESTIGATIONS
As presented in figure 1, flexible nasal endoscopy
revealed a cystic process in the right inferior and
middle nasal passage; there was no further clinical
pathology.
CT scans (see figure 2) of the paranasal sinuses
showed a completely obliterated right maxillary
sinus with a ballooning effect, formally of the
medial and lateral osseous boundary with suspi-
cious pressure atrophy (differential diagnosis:
destruction) and a hardly identifiable middle and
inferior conchae.
DIFFERENTIAL DIAGNOSIS
1. Odontogenic sinusitis: Up to 40% of chronic
bacterial maxillary sinus infections are attribut-
ed to a dental source; the CT scan would de-
mask evidence of periapical lucencies.1
2. Late- stage inverted papilloma (IP): IP can pres-
ent with unilateral nasal obstruction and rhinor-
rhoea; in the CT scan, IP in 28% presents in the
maxillary sinus, is mostly solid and can cause
bony erosions.2
3. Maxillary mucocele (MM): The MM is a slow-
growing, mucous- containing lesion lined with
epithelium that occurs due to ductal obstruc-
tion and can cause irregular osteolysis in the CT
scan.3
TREATMENT AND TIMELINE
Transnasal functional endoscopic sinus surgery
(FESS) under general anaesthesia was performed,
and histopathology revealed mucosa with chronic
sinusitis and a polypoid atypic proliferation. Due
to inconsistent results, pathology was referred and
stated the diagnosis of AM.
As seen in figure 3, a postoperative CT scan
showed a significant tumour regression with an
increasing ossification of the lateral maxillary sinus
in comparison with the primary CT (see figure 2).
Complementary MRI only showed cystic mucosal
areas and a peripheral mucosal enrichment, without
any signs of malignancy.
Our tumour board recommended a transvestib-
ular maxillary sinus surgery, with an intraoperative
histological examination and possibly resection of
the sinus floor including teeth extraction.
FESS was performed combined with trans-
vestibular partial mucosal resection of the right
ethmoidal cells and the right maxillary sinus—
further parts of the posterior wall of the maxil-
lary sinus. No tooth extraction was necessary. A
histological in sano resection was being achieved,
without a radical maxillectomy. No intraopera-
tive or postoperative complications disturbed the
patient’s recovery.
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2FiedlerLS, WunschA. BMJ Case Rep 2021;14:e241487. doi:10.1136/bcr-2020-241487
Case report
Figure 1 Transnasal flexible endoscopy with a cystic formation in the
middle nasal passage.
Figure 2 Preoperative coronary (left) and axial (right) CT scan with a
tumorous mass in the right maxillary sinus and pressure atrophy of the
lateral sinus wall (erosion).
Figure 3 Coronary (left) and axial (right) CT scans after functional
endoscopic sinus surgery.
Learning points
►Symptoms of sinusitis can mask a broad range of differential
diagnoses.
►In paranasal sinus tumours, due to radiographic findings a
definitive and precise diagnosis is not predictable; therefore,
obtaining of histology is essential.
►Ameloblastoma involving the maxillary sinus is rare and
shows a high recurrence rate.
►The gold standard of treatment for ameloblastoma is
radical resection, but conservative treatment options can be
discussed to minimise postoperative impairments probably
due to the difficulty to perform radical surgery at that site
►In conservative treatment options of ameloblastoma in the
maxillary sinus, a higher recurrence rate should be discussed
with the patient
OUTCOME AND FOLLOW-UP
The patient showed no functional problems or signs of recur-
rence within a follow- up period of 4 months. Radiographic MRI
scans showed a normal mucosal lining within the right maxillary
sinus, further regressive osseous defects of the lateral sinus wall.
DISCUSSION
AM is a histologically benign tumour and accounts for 1% of
all head and neck tumours.4 Although histopathologically AM is
classified as benign, residual postoperative tissue, for example,
in the maxillary sinus can affect the skull base and lead to fatal
complications. AM is 80% present within the mandible and 20%
within the maxilla. Most AM originate from gnathic tissue,5 and
a subset can occur from the epithelial lining of the maxillary
sinus.6 Listed in the WHO classification of odontogenic tumours
from 2017, AM can present as a unicystic type, extraosseous/
peripheral or metastasising.7 Given the fact that AM can arise
from follicular tissue of a upper third molar, we tried to organise
preoperative panoramic X- rays of the patient, which were not
existent.
The diagnosis of maxillary AM is commonly at a late stage8
and can be covered by symptoms of recurrent sinusitis. In our
case, a 38- year- old teacher, suffering from a blocked nose and
typical symptoms of recurrent sinusitis for over 2 months, was
planned for FESS. Preoperative planning led to a CT scan,
demasking a cystic lesion of the right maxillary sinus with suspi-
cious lateral pressure atrophy.
We performed FESS under general anaesthesia; this histo-
pathologically revealed chronic sinusitis and a polypoid atypic
proliferation. Due to inconsistent results, pathology was referred
and stated the diagnosis of AM.
AM slowly invades adjacent structures and tissue in a painless
and asymptomatic behaviour.5 This fact explains the late onset
of symptoms in our patient. While the duration of symptoms to
therapy was short, the extent and atrophy of bone were dispro-
portionally high.
Due to anatomical structures and the adjacent structures of
the maxilla, AM due to its invasion5 is further associated with
an aggressive course and can extend to the skull base.9 Given
the fact that recurrence is highly possible, the chosen main-
stay course of resection is radical.8 The current gold standard
of treatment for maxillary sinus AM is wide local excision with
clear margins and immediate reconstruction. Almost AM in this
region is treated with open partial maxillectomy10; further radi-
ation can be a feasible therapy option.8
In our case, we discussed the pathway with our patient—a
well- informed teacher—and decided to minimise the radicality
and chose a combined approach over FESS and transvestibular
partial mucosal resection of the right ethmoidal cells and the
right maxillary sinus. We further resected parts of the poste-
rior wall of the maxillary sinus. Given the fact that in our case
AM histologically seemed to be extraosseous and no radicular
involvement was seen, we decided not to extract teeth within
the operation. We aimed for a minimal functional impair and a
maximum outcome.
Risk factors of recurrence are discussed controversially; Yang
et al identified soft tissue or maxillary sinus involvement as statis-
tical risk factors of AM recurrence in 890 patients.9 Therefore
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FiedlerLS, WunschA. BMJ Case Rep 2021;14:e241487. doi:10.1136/bcr-2020-241487
Case report
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we incorporated the patient into our oncological after- care
programme. Here, we will periodically perform CT and MRI
scans and endoscopically check the nasal and paranasal sinuses
for signs of recurrence. Follow- up should be done for at least
10 years, as recurrence is possible after a long period of time,
especially in this case because there was no radical resection
performed. Since the patient is not that old, MRI is performed
every 6 months (last postsurgical control was also an MRI) and
could if necessary, for example, in unclear cases be comple-
mented by a CT. Clinical follow- up is also done and recalled
every 6 months.
Acknowledgements I acknowledge my chief of department Dr Schäfer and the
maxillofacial surgeon Dr Engels who operated on the patient and supported the
decision to publish the case.
Contributors LSF was involved in writing, selection of publications, editing and
revisions I and II. AW was involved in writing, review, editing and revision I.
Funding The authors have not declared a specific grant for this research from any
funding agency in the public, commercial or not- for- profit sectors.
Disclaimer Case reports provide a valuable learning resource for the scientific
community and can indicate areas of interest for future research. They should not be
used in isolation to guide treatment choices or public health policy.
Competing interests None declared.
Patient consent for publication Not required.
Provenance and peer review Not commissioned; externally peer reviewed.
ORCID iD
Lukas SFiedler http:// orcid. org/ 0000- 0001- 9319- 8260
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