2425
Views & Citations1425
Likes & Shares
Introduction: The
nasolabial cyst is a rare non-odontogenic, soft-tissue characterized by its
extra osseous location in the nasal alar region. Published African data of this
condition are not easily available and according to the literature, most
patients affected are women. The patient presenting this condition is a boy
born and living in the Democratic Republic of Congo. Its pathogenesis remains
uncertain. The diagnosis is essentially based on clinical findings. Bimanual
palpation and imaging findings help to confirm the diagnostic.
Symptoms
and clinical findings: This publication reports a case of an 18
year old boy with an 11 months’ history of a non-painful swelling of the left
nasolabial region which has progressively increased in size and caused facial
deformity without facial numbness. Physical extra oral examination revealed on
the left nasal ala a mass soft, fluctuant, well circumscribed, non-tender and
mobile.
Diagnostic: The
clinical presentation suggested a nasolabial cyst. The patient underwent a
nasal marsupialisation under local anesthesia via the sub labial approach. The
diagnostic of the nasolabial cyst was confirmed by histopathology.
Conclusion: Despite
the rarity of published data of nasolabial cyst from the African region, the
disease exists and every clinician should recognize the clinical
characteristics of this lesion. The cure of this cyst can be obtained
definitely in poor settings by nasal marsupialisation under local anesthesia
which requires minimal equipment. The objective of this paper is to review the
literature, discuss the epidemiology, the diagnosis and the management of this
condition in poor settings.
Keywords:
Nasolabial cyst, Non-odontogenic cyst, Transnasal marsupialization
Abbreviations: CT: Computerized
Tomography; MRI: Magnetic Resonance Imaging; ENT: Ear, Nose and Throat
INTRODUCTION
The nasolabial cyst is
an uncommon non-odontogenic masses that originate from maxillofacial
soft-tissue in the nasal alar region. The first documentation of nasolabial
cyst was done by Zuckerkandl in 1882 [1].
According to recent
reviews on this condition, the nasolabial cyst is rarely diagnosed in Western
countries but may be more frequent in other regions, e.g. Eastern Asia [2].
African statistics including Congolese’s are not available.
The age of detection
ranges from 12 to 75 years old; the nasolabial cysts predominantly affect
people aged between 12 to 75; however, a peak incidence has been noted in the
fourth and fifth decades of life, with a female predilection of nearly 3:1 [3].
Our patient is a young 18 years old boy who had a swelling contiguous to the
nose with fullness in the canine fossa and the nasal floor. The swelling causes
the deformity of the nasolabial sulcus and the elevation of the ala nasi on the
left. The nasolabial cyst is a fluctuant mass best palpated bimanually. It is
lined by pseudostratified ciliated or non-ciliated columnar epithelium with
goblet cells. To diagnose this condition, the literature suggests, in addition
to clinical findings, to do imaging such CT scan and MRI which are not often
available in some area of Africa and if present, they are expensive. Many techniques of efficient surgical excision have been
described in the literature such endoscopic approach but they are costly and
are not available in many places of low-income countries.
In this report, we have described one the
rare cases of nasolabial cyst published from Democratic Republic of Congo with
its clinical features, diagnosis and treatment.
CASE REPORT
Patient information
The patient described in the present work has
signed informed consent.
An 18 years old boy reported on November 2017
in ENT department of our hospital with an 11 months’ history of a non-painful
swelling of the left nasolabial region which has progressively increased in
size and caused facial deformity without facial numbness. No congenital
deformity was noted. No history of trauma and fractures were reported.
This clinical presentation has left the young
boy living in anxiety because of the facial deformity and the fear of getting
incurable cancer. School mates and friends have been laughing at him and some
relatives have been said that someone cast to him a spell so he has become a
curse in the family. For this reason, the parents took him to the traditional
healer in order to chase away the spell and got purified, unfortunately without
success.
Clinical findings
Physical extra oral examination revealed
asymmetry of the face and the deformation of the left nasolabial sulcus and
elevation of the ala nasi on the left side.
On examination, there is a mass 7 × 6 cm
occupying the region between the left upper alveolar ridges to the inferior
border of the left zygomatic arch. The mass is soft, fluctuant, well
circumscribed, non-tender and mobile over the underlying structures. Unilateral
elevation of the nasal ala to the left side is noted.
Physical intraoral examination showed no
trismus, a smooth mucosal covered mass in the gingival labial sulcus is seen at
the upper left side displacing the upper left canine tooth. The mass is
rounded, non-tender not bleed in contact and clearly circumscribed. The hard
palate is normal. No bulge of the soft palate is noted. The gloss alveolar
sulcus is free. No pathology is seen in glosso-alveolar and gingiva-alveolar
sulci.
Diagnostic
assessment
After transnasal
marsupialisation
Therapeutic
intervention: A
diagnosis of the nasolabial cyst was made and nasal marsupialisation excision
via sub labial approach under local anaesthesia was successfully done.
Follow-up and
outcomes: No
postoperative complications were seen and no lesion recurrence was noted after
12 months of follow up.
DISCUSSION
Nasolabial cyst in an uncommon lesion located
near the alar cartilage extending into the superior nasolabial groove and the
floor of the nasal vestibule. It is classified as a non-odontogenic, extra osseous
cyst and is usually located in the area of the nasolabial sulcus, just below
the ala nasi. It accounts for approximately 7% of maxillary cysts and is
unilateral in 90% of cases and bilateral lesions have been reported in the
literature. It represents about 0.7% of all cysts in the maxillofacial region
and 2.5% of non-odontogenic cysts [2,3].
This case illustrates the classical clinical
presentation of a nasolabial cyst. The clinical features described in many
publications were observed in this case especially the effacement of the
nasolabial sulcus and the elevation of the ala nasi. According to the
literature some patients can be asymptomatic but most have at least one of the
three following partial or complete nasal blockage, well-circumscribed swelling
and local pain (all were found on our patient). Nevertheless, in order to
appreciate the extension of the lesion and the bone erosion, CT scan and MRI
were not done due to unavailability in our town and financial constraints. It
was also difficult to compare our data to others because the published data
about the same condition are not easily available.
Pathogenesis
The
pathogenesis of nasolabial cysts is still uncertain. Two theories have been
suggested to explain the origin of nasolabial cyst:
1. Klestadt
in 1913 suggested that they arise from trapped epithelium at the point where
the maxillary, medial nasal and lateral nasal processes fuse which become
inclusion cyst (fissural cyst). However, a lack of evidence to support the idea
of embryonic epithelial entrapment in this location prompted many researchers
to discard this hypothesis.
2. Bruggeman
in 1920 had suggested that nasolabial cysts develop from remnants of the
embryonic nasolacrimal ducts (developmental cyst). This theory is supported by
the fact that the nasolacrimal ducts are lined with pseudostratified columnar
epithelium, which is the type of epithelium found in the nasolabial cyst cavity.
Currently, it is the most widely accepted theory [4].
Diagnosis
Symptoms and signs: The nasolabial cyst is usually
asymptomatic. The patient presents only when the cyst becomes infected or when
it causes unilateral fullness in the nasolabial region. Patients initially
noticed a fullness in the nasolabial region before it becomes symptomatic. Due
to the particular presentation and location of these lesions, their diagnosis
is almost exclusively clinical. The most common sign is enlargement causing
facial asymmetry due to the displacement of the upper lip, with an elevation of
the ala nasi and effacement of the nasolabial sulcus. Local pain, nasal
obstruction, and concomitant infection which can lead to abrupt enlargement of
the lesion may also be present. Occasionally in late presentation, it can
present with nasal obstruction when it pushes on the inferior turbinate causing
it to medialize [1-3].
On inspection,
nasolabial cyst appears to be either normal pink or bluish in color. The cyst
is best palpated bimanually with a finger in the floor of the nose and other in
the labial sulcus. The cyst appears underneath the ala nasi as a painless
fluctuant swelling extending laterally into the cheeks, often obliterating the
nasolabial sulcus, and extending anteriorly into the lip and mucobuccal
vestibule [2,3].
Imaging: Periapical
radiographs, nasolabial cysts may present as a radiolucent area in the apical
region of the maxillary incisors. Standard occlusal views show posterior
displacement of the radiopaque line corresponding to the bony margins of the
anterior nasal aperture.
In the absence of radiographic findings and
when a more precise analysis of the borders of the lesion is required, CT SCAN
is the imaging modality of choice. CT scans usually reveal a homogeneous,
well-delimited cystic lesion in the lateral nasal region cystic lesion, with no
contrast uptake. Larger lesions may be associated with bone remodeling of the
underlying maxilla. CT is able to demonstrate soft tissue nature as well as
bony involvement. As the cyst is benign there is no bony erosion other than
expansible lesion causing thinning of the bone [5].
Ultrasonography does not offer much other
than to reveal the cystic nature of these lesions, for example, well
circumscribed, rounded or oval shapes and anechoic fluid-filled masses in the
nasolabial sulcus region [5,6]. Magnetic Resonance Imaging (MRI) shows the
characteristics of the fluid in T1 (low intense) and T2 (bright) views [2,5].
Histopathology
Histopathological examination reveals ciliated
pseudostratified columnar epithelium and occasionally, stratified squamous
epithelium. In a scanning electron microscopy study of the inner surface of
nasolabial cysts, non-ciliated columnar epithelium with basal cells and goblet
cells is found [3,4].
Differential
diagnosis
Differential diagnosis of the nasolabial cyst
includes:
a. Odontogenic cyst: It
originates from tissues involved in tooth development. So careful examination
will show evidence of non-vital tooth with radiolucency.
b. Dentigerous cyst: most
common sites are mandibular third molar and maxillary third molar, large cysts
tend to expand the outer plate (usually buccally).
c.
Dermoid
or epidermoid cyst: As opposed to the normal pink or bluish
coloration of a nasolabial cyst, this cyst is yellow in color.
d. Fibrous-osseous disease:
Painful, hard, bone is replaced by fibrous tissue [3,7].
TREATMENT
Treatment is aimed to prevent infection, to
improve a cosmetic deformity and to establish a histopathological diagnosis.
The current treatment of nasolabial cyst is
complete excision:
a. Surgical
enucleation is easily achieved via a transoral sublabial approach.
b. Transnasal
marsupialisation of the nasolabial cyst which opens into the nasal cavity has
reported no recurrence of the cyst. It is the technique applied to our patient.
It is 12 months since the excision was done no recurrence has been reported
[8].
c.
Recently, the alternative transnasal
route was proposed by some authors: Endoscopic approach extends the nasal floor
to the former cystic cavity and thus prepares an air-containing sinus. This
technique appears to allow sufficient drainage of the new sinus and there were
no signs of cyst recurrence [9-11].
d. Other
modes of treatment that had been described are simple aspiration, injections
with a sclerosing agent, destruction by cautery, needle aspiration and incision
and drainage. However, these methods have been found associated with high
recurrence rates [8,11].
CONCLUSION
From the literature and the present case, the
diagnosis is based on clinical findings. Despite the rarity of the nasolabial
cyst, the general practitioners, ENT specialists and dental practitioners
should recognize the key features of these lesions in order to be able to
distinguish them from the odontogenic origin and enable appropriate treatment.
Therefore, a diagnostic hypothesis of nasolabial cyst should be based on
clinical examination and imaging; and histopathology is necessary to confirm
the diagnosis. CT and MRI are good to make a diagnosis but clinical examination
can be enough to get a diagnosis of nasolabial cyst in poor settings. Once the
diagnosis is established, the optimal treatment consists of complete excision
using sophisticated equipment but nasal marsupialisation can guarantee good
prognosis and rare recurrences.
DECLARATIONS
Ethics approval and
consent to participate
Not applicable
Consent for publication
Written informed consent for publication of
his clinical details and clinical images was obtained from the patient.
Availability of data
and materials
The datasets used and analyzed during the
current study are available from the corresponding author on reasonable
request.
Competing interests
The authors declare that they have no conflicts
of interest.
Funding
This study received no financial support.
Acknowledgement
We are grateful to Dr. Jeef Mutambala, Mrs
Namushukuru Yvette and Mbindule Eugénie for helpful advice regarding data
collecting and assist in the preparation of the manuscript.
Author’s
contributions
1. Nzanzu Kikuhe
collected the data and wrote the initial draft of the manuscript. He also
performed the operation and collected the data.
2. Sekabuhoro Safari was
involved in the preoperative and postoperative evaluation of the patient.
3. Munyaneza Sekimonyo Claude was
involved in the preoperative and postoperative evaluation of the patient.
4. Emily Kakande
contributed to analysis and interpretation of the data.
5. Justine Namwagala
reviewed the final manuscript.
1.
Toribio Y, Roehrl MH
(2011) The nasolabial cyst: A non-odontogenic oral cyst related to nasolacrimal
duct epithelium. Arch Pathol Lab Med 135: 1499-1450.
2.
Cebi IT, Karatas A,
Yuce T, Salvız M, Kocak A, et al. (2016) Bilateral nasolabial cyst as a rare
case report. Turk Arch Otorhinolaryngol 54: 79-81.
3.
Sheikh AB, Chin OY,
Fang CH, Liu JK, Baredes S, et al. (2016) Nasolabial cysts: A systematic review
of 311 cases. Laryngoscope 126: 60-66.
4.
Friedrich RE, Scheuer
HA, Assaf AT, Zustin J (2012) Nasolabial cyst: Case report with respect to
immunohistochemical findings. In Vivo 26: 453-458.
5.
Aquilino RN, Bazzo VJ,
Faria RJA, Eid NLM, Bóscolo FN (2008) Nasolabial cyst: Presentation of a
clinical case with CT and MR images. Braz J Otorhinolaryngol 74: 467-471.
6.
Acar AH, Yolcu U,
Asutay F (2014) Is ultrasonography useful in the diagnosis of nasolabial cyst?
Case Rep Dent 2014: 1-3.
7.
Goyal S, Sharma J,
Sharma N (2014) A case report on nasolabial cyst. Int J Cancer Ther Oncol 2:
020311.
8.
Yuen HW, Julian CL,
Samuel CY (2007) Nasolabial cysts: Clinical features, diagnosis and treatment J
Oral Maxillofac Surg 45: 293-297.
9.
Goyal S, Sharma J,
Sharma N (2014) A case report on nasolabial cyst. Int J Cancer Ther Oncol 2:
02031.
10.
Ozer S, Cabbarzade C,
Ogretmenoglu O (2013) A new transnasal approach to nasolabial cyst: Endoscopic
excision of nasolabial cyst. J Craniofac Surg 24: 1748-1749.
11. Imre A, Pinar E,
Calli C, Oncel S (2012) Endonasal endoscopic approach for recurrent nasolabial
cyst. J Craniofac Surg 23: 438-440.
QUICK LINKS
- SUBMIT MANUSCRIPT
- RECOMMEND THE JOURNAL
-
SUBSCRIBE FOR ALERTS
RELATED JOURNALS
- BioMed Research Journal (ISSN:2578-8892)
- Journal of Nursing and Occupational Health (ISSN: 2640-0845)
- International Journal of Medical and Clinical Imaging (ISSN:2573-1084)
- Advance Research on Alzheimers and Parkinsons Disease
- Journal of Infectious Diseases and Research (ISSN: 2688-6537)
- Archive of Obstetrics Gynecology and Reproductive Medicine (ISSN:2640-2297)
- Journal of Oral Health and Dentistry (ISSN: 2638-499X)