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Case Report
Aggressive nasopalatine duct cyst with
complete destruction of palatine bone
D. Sankar, Veerabahu Muthusubramanian, J. A. Nathan, Ravi Sankar Nutalapati,
Yasmin Mary Jose, Y. Naren kumar

Department of OMFS, ABSTRACT


Ragas Dental College and
Nasopalatine duct cyst is the nonodontogenic developmental cyst, frequently occurring in the midline of the
Hospital, Chennai,
Tamil Nadu, India
anterior maxillary region. The clinical presentation of the cyst is often varied and presents a diagnostic difficulty
and frequently misdiagnosed as developmental or inflammatory odontogenic cystic lesion. This paper represents
Address for correspondence: a large infected nasopalatine duct cyst presenting with complete destruction of anterior palate and pyriform rim.
Dr. D. Sankar,
E‑mail: drsankarmds@gmail.
com

Received : 06-04-16
Review completed : 28-04-16
Accepted : 06-05-16 KEY WORDS: Cone beam computed tomography, enucleation, nasopalatine duct cyst

N asopalatine canal develops at the junction of the primary


palate and the secondary palate. It communicates the nasal
floor with the oral cavity. In general, the epithelial lining in the
weeks. History revealed recurrent episodes of pain and swelling
and spontaneous remission following nasal discharge. On
examination, the patient had a well‑defined, fluctuant swelling
nasopalatine canal will degenerate; however, in some patients, it obliterating the labial vestibule from 12 to 22. The swelling
persists and proliferates to form a cyst in the anterior midpalatal also extended in the midpalatal region between two central
region which is called as nasopalatine duct cyst or incisive canal incisors posteriorly up to the premolar level [Figure 1]. The
cyst. Nasopalatine cyst is often misdiagnosed as inflammatory central incisor exhibited mild Grade 1 mobility buccopalatally
cyst originating from the incisor teeth due to the proximity of without any movement in the corono‑apical region. Both
the cyst to the incisors. The cystic lesion in the anterior maxilla central incisors were tender on palpation. On vitality test, the
is most commonly radicular cyst followed by dentigerous cyst central incisors were found nonvital. Patient had poor oral
followed by keratocystic odontogenic tumor.[1] It constitutes hygiene and inflamed gingiva with 5 mm of gingival pocket in
<1% to 33% (average 12%) of all the cysts of jaw,[1‑4] but it is one both central incisors. Patient had generalized bleeding gums.
of the most commonly reported nonodontogenic developmental On aspiration, straw‑colored fluid with mixture of blood was
cysts of the jaw. Although nasopalatine duct cyst can occur at any found [Figure 2]. There was no lymphadenopathy found. The
age, the incidence is more common in the late adults between periapical radiograph showed large triangular‑shaped radiolucent
40 and 60 years age group.[5,6] However, it can present in young lesion measuring about 2 cm diameter was present between the
patients[5,7] also. In this case report, we are presenting the classical two maxillary central incisors [Figure 3]. The lesion had a thin
features of nasopalatine duct cyst which were found in the clinical, radio‑opaque margin with displacement of both the roots in a
radiographic as well as on histopathological examination. distal direction [Figure 4]. Mild root resorption was observed
in 21 root and the lesions also involved the periapical region of
Case Report the two lateral incisors. The central incisors had bone only on
the cervical region, and the remaining portion was completely
A 40‑year‑old male patient came with the complaint of pain in the radiolucent lesion. The nasal septum was found in the
and swelling in the anterior vestibular region for the past 2
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DOI: How to cite this article: Sankar D, Muthusubramanian V, Nathan JA, Nutalapati
RS, Jose YM, kumar YN. Aggressive nasopalatine duct cyst with complete
10.4103/0975-7406.191956
destruction of palatine bone. J Pharm Bioall Sci 2016;8:S185-8.

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Sankar, et al.: Aggressive nasopalatine duct cyst

Figure 2: Aspiration content

Figure 1: Preoperative intraoral swelling

Figure 4: Orthopantamograph showing distal displacement roots of


upper central incisors
a b
Figure 3: Preoperative (a) intraoral periapical radiograph, (b) occlusal
and informed consent was obtained. As the cystic wall was
radiographs
found attached to the nasal mucosa and palatal mucosa, both
labial and palatal approach were used [Figure 6]. The cyst was
middle of the radiolucent lesion. Occlusal view radiograph carefully dissected and enucleated, without breaching the nasal
revealed an oval radiolucent lesion from the central incisor mucous membrane or the palatal mucosa. Intraoperatively, the
to the 2nd premolar region in the midpalatal region measuring cyst was found to be a capsuled lesion which was adherent in
about 3 cm × 2 cm lesion. Due to the proximity of the lesion the nasopalatine region of palatal mucosa but was not attached
to the nasal floor, cone‑beam computed tomography (CBCT) to the root or periodontal ligament. The flap was closed, and
scan was taken to plan for the surgical procedure which clearly palatal acrylic plate was placed to support the palatal mucosa.
showed the complete breach of both the nasal floor and hard Postoperative period was uneventful. Histopathologically, it
palate with hollowing in the anterior maxillary region [Figure 5]. was found to have fibrous capsule with stratified squamous
The patient’s medical history was noncontributory. Based on epithelial lining, and final diagnosis of nasopalatine duct cyst
the clinical and radiographic findings, provisional diagnosis of was confirmed [Figure 7].
nasopalatine duct cyst was made.
Discussion
The patient was advised about the nature and severity of the
lesion, and the complete bone loss of anterior palatine bone was Nasopalatine duct canal develops as a right and left side canal
explained. The significance of surgical enucleation of the lesion but generally fuses into single canal. However, three anatomical
was explained to the patient and also advised for bone graft variations have been reported; wherein, it sometimes appears
placement. The patient gave consent for the cyst enucleation as two parallel canals or single canal or a Y‑shaped canal.[8,9]
but was not willing for bone grafting procedure. Before the Depending on the anatomical structure, the nasopalatine cyst
surgery, root canal treatment of the involved teeth was done, appears as a midline cyst or paramedian cyst when it involves
and complete scaling was done to improve the oral hygiene. one side of the canal only. However, midline nasopalatine duct
cyst is more common than the other types (Stafne 1969).[6]
Before surgery, all preliminary investigations were done, and
the results were within normal limit. Due to the mild mobility The etiology of nasopalatine duct cyst is idiopathic or reactive.
of the central incisors, splinting of the teeth with arch bar was It has been postulated that localized trauma such as previous
done before the surgical procedure. The possibility of tooth endodontic treatment, implant placement or infection or
avulsion or extractions of incisors were explained to the patient, mucous retention may initiate the cellular proliferation and may

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Sankar, et al.: Aggressive nasopalatine duct cyst

Nasopalatine duct cyst has been reported to be more prevalent


in females.[5] It may appear as extrabony incisive papilla cyst
or intrabony lesion. It appears as either asymptomatic which
is recognized on routine imaging or may present with pain or
swelling or with other symptoms such as burning sensation,
tooth displacement, nasal obstruction, or discharge into the
nasal cavity or oral cavity.[5] The size of the swelling is variable in
a b size but most often it is <2 cm in diameter. However, occasional
Figure 5: (a and b) Cone beam computed tomography images of the cases of large and aggressive lesion up to 52 mm have been
lesion showing complete anterior palatine bone loss reported with complete destruction of labial and palatal bony
plates.[5,16,17]

When the nasopalatine duct cyst present at higher level, there


is not much effect over the tooth. The nasopalatine duct cyst
when it is present at lower level always displaces the tooth and
often considered characteristic of the lesion. The tooth vitality
associated with the cyst is variable.[1] Most literature reports the
a b presence of lamina dura and maintenance of vitality. However,
Figure 6: Intraoperative picture showing attachment of the cystic wall it has been reported in association with nonvital tooth and
with (a) nasal mucosa, (b) palatal mucosa periodontally compromised tooth. In our case, both the incisors
were nonvital, and the patient had chronic periodontitis with
poor oral hygiene. Root resorption is uncommon; however,
Shear and Speight have reported cases with root resorption.[6,13]

Radiographically, it appears as a round, ovoid, pear‑shaped, or


heart‑shaped radiolucent lesion in the middle of the palate. It
rarely causes root resorption. Bone expansion is uncommon,
and usually, it causes resorption of bone. In our case, the
cyst completely eroded the palatal bone and the nasal floor,
and the swelling was fluctuant in nature. Nasopalatine duct
cyst should be differentiated from the normal nasopalatine
fossa. The average size of the normal nasopalatine fossa is
3 mm in anteroposterior, transverse, and in vertical direction.
However, Shear and Speight have reported that the mean size
of the nasopalatine fossa was as high as 10.19 + 3.24 mm
anteroposteriorly and 4.79 + 1.33 mm in width. It is advised
that if the radiographic size of nasopalatine fossa was <10 mm
Figure 7: Histopathological picture showing stratified squamous without any signs and symptoms, it should be re‑radiographed
epithelium (H and E, original magnification, ×10) periodically. However, Bodin, Isaacson, and Julin advised for
surgical exploration if the size of the radiolucency exceeds 8 mm,
give rise to nasopalatine duct cyst. It has also been reported to or if it shows asymmetric bulging.[6] Aspiration of the radiolucent
present in association with impacted mesiodens.[5,10‑13] It is not lesion will help to differentiate a normal nasopalatine fossa
been associated with any habits such as smoking or alcoholism.[4] from the cyst. CBCT is useful when planning for the surgical
In our study, it appears to be secondary to chronic periodontitis enucleation of the lesion as it clearly shows the proximity of
or nonvital teeth. the lesion with the nasal floor and the posterior extent in the
hard palate. The differential diagnosis of the lesions includes
The cyst arises from the epithelial lining of nasopalatine duct odontogenic cysts and tumors such as dentigerous cyst,
or from the vomer nasal organ of Jacobson or mucous glands lateral periodontal cyst, cystic ameloblastoma, keratocystic
present in the nasal mucosa.[6] Histologically, the nasopalatine odontogenic tumors, and periapical granuloma.[1]
duct exhibits different epithelial cells in different region.
The epithelium close to the nasal floor has ciliated columnar The treatment of nasopalatine duct cyst is enucleation of
epithelium, and the one closer to the oral cavity has squamous the lesion. However, marsupialization is considered in large
epithelium and the one in the middle has cuboidal epithelium. lesions followed by second stage enucleation can be done.
It has also been reported containing nerve fibers and melanin Palatal approach has been reported frequently in most of the
or lipofuscin pigments.[4,14,15] Hence, the nasopalatine cyst also literature. Autogenous bone grafting can be placed. Recurrence
exhibits the mixed nature of the epithelial lining. In our case, is uncommon following enucleation; however, recurrence rate of
the cystic lining was of squamous epithelial type. 11–30% has been reported in the literature.[1,4] In our study, we

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Sankar, et al.: Aggressive nasopalatine duct cyst

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Financial support and sponsorship its corrective therapy. A case report. Oral Surg Oral Med Oral Pathol
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