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Oral Surgery ISSN 1752-2471

CASE REPORT

Nasolabial cyst: a case report


I. Zografos1, L. Podaropoulos1 , E. Malliou2 & K. I. Tosios3
1
Department of Oral and Maxillofacial Surgery, School of Dentistry, National and Kapodistrian University of Athens, Athens, Greece
2
Private Practice, Athens, Greece
3
Department of Oral Pathology, School of Dentistry, National and Kapodistrian University of Athens, Athens, Greece

Key words: Abstract


Klestadt cyst, nasoalveolar cyst, nasolabial
cyst, non-odontogenic cyst Nasolabial cysts are rare non-odontogenic cysts of soft tissue that usually
present as asymptomatic swellings beneath the ala of the nose. Their
Correspondence to: pathogenesis is uncertain. The presence of the lesion is perceived
I Zografos clinically and by the aid of radiological imaging. The usual treatment is
Department of Oral and Maxillofacial Surgery
surgical excision. Nasolabial cysts, when infected, are painful and
School of Dentistry
mistreated occasionally as odontogenic defects. This article reports a case
National and Kapodistrian University of
Athens of a nasolabial cyst that was initially diagnosed and treated as a
Aggelidou 10-12 periapical abscess, which lead to the loss of a tooth. Complete
P. Faliro 17561 enucleation of the cyst was performed under local anaesthesia and its
Athens identity was confirmed by histopathology. Follow-up over a year
Greece revealed no evidence of recurrence.
Tel: +302109828103
Fax: +302107461267
email: ioannis_zografos@hotmail.com

Accepted: 22 April 2018

doi:10.1111/ors.12365

Introduction cause facial asymmetry and deformity8,10. It mea-


sures 1 to 5 cm in diameter11. Nasal obstruction may
The nasolabial cyst is an uncommon, non-odonto- ensue, and pain follows infection of the cyst12,13.
genic developmental cyst with an incidence esti- Diagnosis is established by microscopical examina-
mated to 0.7% of all maxillary and mandibular tion. A combination of pseudostratified squamous,
cysts1,2. It is located in the soft tissues of the upper stratified squamous, respiratory and cuboidal epithe-
lip, inferior to the nasal alar region3. Its first descrip- lium may be seen, and inflammatory cells are pre-
tion is credited to Emil Zuckerkandl in 18934. Naso- sent8. Goblet cells are found in 55.6%2 to 67%14 of
labial cyst is, also, referred as nasoalveolar cyst, but cases.
this name should be abandoned as it is a soft tissue We present a case of an infected nasolabial cyst in
cyst and the alveolus is not involved. Less common a 60-year old male that was initially diagnosed and
names used in the past are nasovestibular cyst, treated as a periapical abscess.
nasoglobular cyst, subalar cyst and Klestadt’s cyst5,6.
Nasolabial cysts shows a female preponderance of
a ratio ranging between 2.7:1 and 3.7:11,7,8. They Case report
are mostly unilateral, equally affecting both sides1,8, A 60-year old male presented to the general dentist
but bilateral occurrence is reported in slightly more with a chief complaint of a painful swelling in the
than 10%1,8,9. Clinically, the nasolabial cyst presents upper lip. He stated that the lesion was present for
as a slow-growing painless soft tissue swelling of the at least one year and did not give a history of
upper lip elevating the ala of the nose that may trauma, infection or surgical operation in the area.

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Nasolabial cyst Zografos et al.

His medical history was non-contributory. The asso-


ciated teeth were not sensitive to percussion, but the
left upper canine had negative reaction to thermal
vitality tests. An intraoral radiograph did not reveal
any pathologic signs. The case was misdiagnosed as
periapical infection associated with a non-vital upper
left canine, therefore apicectomy with simultaneous
endodontic treatment was performed. During the
procedure pulp necrosis of the tooth was confirmed,
as tissues in the root canal presented with a charac-
teristic black appearance and odour of a necrotic
pulp. Amoxicillin (1000 mg) and metronidazole
(500 mg) were prescribed twice daily for 8 days. This
broad spectrum antibiotic regimen was prescribed by
the referring general dental practitioner due to the
severity and persistence of the symptoms. However,
the patient was not relieved from his symptoms and
the relevant canine was extracted, since it was con-
sidered responsible for the persistence symptoms. As
no improvement followed patient’s condition he was
Figure 2 Intra-oral pre-operative view of the lesion: a reddish mucosa
referred to our team for further investigation.
is detected in the buccal area of the extraction site of the upper left
Clinical examination confirmed the presence of a canine.
slight facial asymmetry and a deformity in the left
nasolabial region (Fig. 1). On extraoral palpation a
round, tender and diffuse swelling was located conventional CT of the maxilla was performed
underneath the left ala of the nose. Intraorally, the revealing a homogeneous, well circumscribed round
swelling was covered by reddish mucosa and was soft tissue mass left to the midline, below the alar
soft (Fig. 2). base, protruding and narrowing the entrance of the
The region was radiographically examined by cone left nasal cavity (Fig. 4). The lesion measured
beam computed tomography (CBCT) scan, but 3.4 cm 9 3.0 cm.
no areas of pathology were detected (Fig. 3). A Periapical abcess or radicular cyst was excluded
from the differential diagnosis spectrum as no rele-
vant teeth were left and involved in the region.
Odontogenic cyst of non-inflammatory type was
considered, however, no radiolucency on the radio-
graphs was detected. Clinical diagnosis was directed
to either a soft tissue cyst – such as dermoid, epider-
moid or nasolabial cyst – or a soft tissue neoplasm.
Surgical treatment under local anaesthesia and
intraoral approach was decided (Fig. 5). A sublabial
incision was performed and after the elevation of a
mucoperiosteal flap the cyst was completely removed
without perforating the nasal mucosa, however, the
cystic sac was ruptured. The defect was closed by pri-
mary intention and properly sutured using resorb-
able 3/0 vicryl sutures (MonocrylTM, Ethicon,
Somerville, NJ, USA). A cystic sac, approximately
4 9 1.5 9 0.5 cm was fixed in a 10% neutral buf-
fered formalin solution and submitted for pathologic
examination.
Microscopic examination of 5 lm thick tissue
sections stained with haematoxylin and eosin
Figure 1 Extra-oral pre-operative view of the lesion. showed that the wall of the sac consisted of cellular

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Zografos et al. Nasolabial cyst

Figure 3 Dental CT of the area of the extracted upper left canine showing no evidence of bone pathology in the alveolar ridge.

Figure 5 Full thickness mucoperiostal flap extending from the right


canine to the left first molar exposing the lesion.

Discussion
Figure 4 Conventional CT revealed a circumscribed mass in contact Searching the MEDLINE/Pubmed database under
to the left nasal cavity (red arrows).
the terms “Nasolabial cyst”, “Nasoalveolar cyst”,
“Nasovestibular cyst” and “Klestadt’s cyst” resulted
and vascular connective tissue, lined by ciliated, in 94 published case reports. These case reports fol-
pseudostratified columnar epithelium (Fig. 6). No low different methodologies in imaging, diagnosis
goblet cells were found. The connective tissue pre- and treatment modalities. Nevertheless, the final
sented focal inflammatory infiltration by plasmacytes diagnosis and confirmation of the nature of a lesion
and lymphocytes, mostly subepithelial, as well as should be performed by histological analysis.
haemorrhagic infiltration (Fig. 7). The final diagnosis In the case presented herein, the clinical, radio-
was nasolabial cyst. graphic and microscopic features were consistent
Post-operative healing course was uneventful with nasolabial cyst. The patient noticed the lesion
while follow-up at 1 year with conventional CT for least a year before presentation, however, he
showed no evidence of recurrence (Fig. 8). asked for medical advice only after it became

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© 2018 The British Association of Oral Surgeons and John Wiley & Sons Ltd
Nasolabial cyst Zografos et al.

Figure 6 The wall of the sac consisted of cellular and vascular connective
tissue, lined by ciliated, pseudostartified columnar epithelium.

Figure 8 Medical CT one year post-operatively. No pathology


detected.

tumours. Malignant tumours, in particular mucoepi-


dermoid carcinoma, should also be considered.
Intraoral or panoramic radiographs are not usually
informative in the diagnosis of nasolabial cyst, but
help in excluding intra-osseous lesions. Conventional
CT is the most commonly used imaging tool to detect
a suspected nasolabial cyst and preferred to magnetic
resonance imaging (MRI) due to its lower cost8. In
our case, the CBCT also failed to detect the cyst, as it
depicts only the hard tissues, but it was revealed in
Figure 7 The connective tissue presented focal inflammatory infiltra- the conventional CT. In general, intraoral and
tion, mostly subepithelial. panoramic radigraphs as well as CBCT should not be
considered the imaging method of choice for depict-
ing a nasolabial cyst since they are hard tissue imag-
painful. Following the case from its onset, it is not ing modalities. In case conventional CT fails to detect
clear if the nasolabial cyst caused the loss of the the lesion, an MRI should be considered. In both
vitality of the upper left canine, since no pathology conventional CT and MRI nasolabial cyst presents
was detected at the periapical bone in the intraoral as a non-odontogenic homogeneous circumscribed
radiograph, or it was infected from the adjacent mass, with varying intensity, with or without bony
canine. This commonly ensues in nasolabial cyst9,13 erosion2,15,16. Usually conventional CT is preferred
and may mislead clinician towards a diagnosis of due to its lower cost, however, MRI does not expose
periapical abscess or an odontogenic cyst, that in our the patient to ionising radiation. Ultrasonography
case lead to the loss of a tooth. (USG) is a method often used for detecting soft tissue
Differential diagnosis may include nasopalatine lesions, which may be used intraorally as well. In a
duct cyst or globulomaxillary cyst, however, the case report Acar et al.17 concluded that USG was use-
extraosseous location of the nasopalatine cyst aids ful and adequate when nasolabial cyst is suspected.
diagnosis. The diagnostic spectrum will encompass, USG is fast becoming the modality of choice in
as well, soft tissue cysts, such as dermoid or epider- depicting oral soft tissue lesions due to its inexpen-
moid cyst, furunculosis of the base of the nose, sive and non-ionising nature, giving it an advantage
benign tumours, soft tissue and salivary gland over conventional CT or MRI. On the other hand is

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Zografos et al. Nasolabial cyst

an operator dependent method and its interpretation cases where the lesion was surgically excised and a
is determined by the experience of the user18,19. recurrence rate of 2.53% in cases where the transna-
The etiopathogenesis of nasolabial cysts is debat- sal marsupialisation approach was used. Authors
able. Initially, it was considered as a fissural cyst conclude that overall recurrence rate seems to be
derived by epithelial cells entrapped at the fusion irrelevant of the surgical technique involved8. In the
area of the medial and lateral nasal processes and case presented, the nasal mucosa was not perforated,
the maxillary process5,6. Currently, an origin from but the cyst was not removed intact. However, no
embryonic remnants of the nasolacrimal duct tissue recurrence was found in the 1 year follow-up.
is considered more probable and supported by bilat- Sheikh et al.8 in a recent literature review reported 79
eral occurrence of some cysts2,13,20. However, some articles including 311 patients with diagnosed and con-
cysts may develop due to surgical treatments and firmed nasolabial cysts. Several variables, which were
chronic inflammation21. not consistent in all articles, were included. However,
Therapeutically, injection of sclerozing materials, an effort was made to categorise the data based upon
simple aspiration and destruction by cautery13 have patient demographics, location of the cyst, aetiology,
been tried, but show a high recurrence rate2,10,13,22,23. radiological imaging, histology, management and out-
The most common and effective treatment is complete come. Authors concluded that nasolabial cysts are rare,
surgical excision of the cyst, usually performed intrao- are best defined in conventional CT and MRI and the
rally via a gingivolabial incision, under local or general rates of post-operative swelling, pain and recurrences
anaesthesia8. A transnasal approach of endoscopic are similar in the sublabial and transnasal approach.
marsupialisation of nasolabial cysts is described10 that However, they report inconsistencies between articles
according to some8,13,24, but not all25,26 studies shows in the reporting of presentation, cyst characteristics,
good therapeutic results. In a prospective randomised management and complication and they add that the
study27 it was concluded that both methods are effec- heterogeneity of their data may subject their study to
tive. The transnasal marsupialisation has many benefits institutional, geographical and selection bias.
over the conventional intraoral approach, but there is In conclusion, nasolabial cyst, although rare,
no difference with regard to post-operative swelling, should be included in the differential diagnosis in
pain, or overall recurrence rate. Therefore, more stud- cases of sublabial swelling with facial deformity.
ies with longer follow-up periods are needed to esti- Panoramic radiography and CBCT do not give sub-
mate in depth the two different techniques8. A stantial information, therefore, conventional CT or
modification of the above mentioned method that is MRI should be preferred and USG considered. Trans-
suggested is to firstly aspirate the cyst before enucle- nasal marsupialisation or surgical enucleation seems
ation which, according to the authors, exhibits lower to be equally successful in its treatment.
recurrence rates5. Cryosurgery has also been applied as
an alternative treatment method on nasolabial cysts.
Janardhan et al.28 performed cryosurgery, after a sub- Conflict of interest
labial incision, in four patients. The authors suggest The authors confirm that there are no conflicts of
that the application of a cryo probe to the bed of the interest.
cyst after excision is beneficial, especially in cases when
complete cyst wall removal is not a certainty. Further-
more, no complications or recurrence has been Ethical approval
reported with this method. None required.
Care should be taken during surgical excision, as
the tight attachment of the cyst to the nasal mucosa
may lead to perforation of the floor of the nose. References
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