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Nasopalatine duct cyst: a rare case report

Dass, Arjuna; Verma, Hitesha; Punia, RPSb

Egyptian Journal of Oral & Maxillofacial Surgery: April 2013 - Volume 4 - Issue 2 - p 39–40
doi: 10.1097/01.OMX.0000428093.16894.dc
CASE REPORTS
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The nasopalatine duct cyst, also known as a nasopalatine cyst or incisive canal cyst, is a developmental, non-neoplastic cyst that is considered to be the most common nonodontogenic cyst. It is one of many pathological processes that may occur within the jawbones. This article reports the case of a nasopalatine cyst involving the premaxilla and maxilla, along with a discussion on the pathophysiology, clinical presentation, histopathological findings, and therapeutic strategies.

Departments of aOtorhinolaryngology

bPathology, Government Medical College and Hospital, Chandigarh, India

Correspondence to Hitesh Verma, MD, Department of Otorhinolaryngology, Government Medical College and Hospital, 3rd floor, D Block, 160030 Chandigarh, India Tel: +91 0172 2665253 x2309; fax: +91 0172 2608488/+91 0172 2609360; e-mail: hitesh_verma72@yahoo.com

Received March 18, 2012

Accepted February 12, 2013

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Introduction

The nasopalatine cyst (NPC) was described in 1914 by Mayer as a result of a process of the remnant cells derived from the fusion of the primary palate with the first branchial arch 1. However, the etiology is still debatable, as some say it arises from a vestigial organ (nasal–vomer Jacobson’s organ) present in some inferior mammals 2. It is one of the most common nonodontogenic cysts, comprising 10% of jaw cysts and occurring in one of every 100 individuals, with a slight male predilection. It is more common during the fourth and sixth decade of life (mean age being 42.5 years) 3. The cyst development is attributed and triggered by several factors: infection (38%), trauma (16%), minor salivary mucus retention, inflammatory stimulus, spontaneous proliferation of the epithelial remnants present in the nasopalatine duct, and idiopathic origin 4.

The most commonly reported clinical symptom is swelling in the anterior part of the palate. These entities are usually treated with surgical enucleation.

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Case report

A 12-year-old boy presented to the outpatient department of ENT of Government Medical College and Hospital (Chandigarh, India) with a history of multiple swellings in the oral cavity for the past 6 years. To start with, these swellings were small and had increased to the present size. There was no history of trauma, pain, or bleeding. The patient also did not have any difficulty in chewing/swallowing or respiration. Examination of the oral cavity revealed four swellings over the hard palate. Three swelling were sessile and circular in shape, were present on the premaxilla, and had a diameter of 1 cm. The fourth swelling was pedunculated, was attached to the maxilla, and had dimensions of 2×2 cm at the base and 3 cm in length (Fig. 1). These swellings were nontender and did not bleed on touch. We hypothesized the possibility of a benign lesion, most likely papilloma. The patient was administered general anesthesia for excision. The masses were excised from the base with a 5 mm margin, and the edges were cauterized. Histopathological analysis revealed a nasopalatine duct cyst (NPDC) (Fig. 2). After a follow-up period of 6 months, the patient is free from disease.

Fig. 1

Fig. 1

Fig. 2

Fig. 2

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Discussion

The NPDC is a developmental, non-neoplastic cyst that is the most common nonodontogenic cyst of the oral cavity, occurring in about 1% of the population. The stimulus for cyst formation from the epithelial remnants of the nasopalatine canal is uncertain, although trauma and bacterial infection are thought to play a role. It has also been suggested that the mucus glands within the lining may cause cyst formation as a result of mucin secretion 5.

NPC are benign lesions but if left untreated, can lead to abnormal growth. Diagnosis is often difficult and can easily be unnoticed at the early stages. The NPC can be divided into two types: cysts of the incisive canal and those of the incisive papilla, both show the same pattern of growth.

The pattern of development is slow and rarely malignant, but the potential of damaging is higher when the medullary cortex is penetrated 6. The probability of recurrance is low, ranging from 0–2% 7.

According to the literature, NPDCs are present in the premaxilla only 8, whereas we report a case in which they were present both in the maxilla and premaxillary regions.

In this case, the progression of the disease was very slow, without invasion into the underlying bone. In various cases, the swelling is associated with a burning sensation, numbness over the palatal mucosa, and pain as a result of pressure on the nasopalatine nerves. Various combinations of swelling, discharge, and pain may occur. Discharge may be mucoid, in which case the patients describe a salty taste, or it may be purulent and the patients may complain of a foul taste 2. Displacement of teeth is a rare finding. The types of epithelia that line the nasopalatine duct are highly variable, depending on the relative proximity of the nasal and oral cavities. Typically, the most superior parts of the ducts are characterized by a respiratory-type epithelial lining. Moving downward toward the oral cavity, the lining changes to cuboidal epithelium. In the most inferior portions, squamous epithelium is the usual type, whereas in our patient, the cysts were lined by squamous to pseudostratified columnar ciliated to flattened epithelium, with congested vessels and seromucinous glands in the wall. The presence of blood vessel, as seen in our patient, is considered by Abrams et al.1 to be the most important criterion for differentiating NPDC from cysts of other origins. These entities are usually treated with surgical enucleation 9; therefore, in this case, we opted for enucleation of the lesion, although marsupialization can be an option for larger cysts 10, especially when a persistent fistula is a concern.

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Acknowledgements

The authors thank the Director/Principle and Medical Superintendent for allowing us to use hospital data for publication.

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Conflicts of interest

There are no conflicts of interest.

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References

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