133 Address for correspondence: Dr. A Sri Kennath J Arul, No.7A, VOC Street, Alagappan Nagar, Madurai, Tamil Nadu - 625 003, India. E-mail: drkennath@gmail.com Nasolabial cyst: Report of a case A. Sri Kennath J. Arul, Sonika Verma, A. Sri Sennath J. Arul 1 , Rashmika Verma 2 Department of Oral and Maxillofacial Pathology, Best Dental Science College, Madurai, 1 Medical Practitioner, Tamil Nadu, India, 2 Dental Surgeon, Rotorua, New Zealand ABSTRACT The nasolabial cyst is an uncommon non-odontogenic cyst arising in the maxillofacial tissues. This lesion presents in an extraosseous location in the region of the nasolabial fold and can cause swelling in the furrow, alar nose elevation, and upper lip projection. Despite the uncommon occurrence of nasolabial cysts, it is important to recognize the characteristics of this lesion. The paper documents the presentation of nasolabial cyst in a 33 year old woman and discusses considerations related to the diagnosis. Key words: Cyst, diagnosis, nasolabial, non-odontogenic Access this article online Website: www.jofs.in DOI: 10.4103/0975-8844.106212 Quick Response Code: INTRODUCTION The nasolabial cyst (NC) is a rare non-odontogenic cyst originating in the maxillofacial soft tissues. [1] It represents about 0.7% of all cysts in the maxillofacial region, [2] 2.5% of the non-odontogenic cysts. [1]
Many authors believe that its prevalence is actually higher than that presented in the literature; however, due to misdiagnosis, indexes remain low. [1] These cysts, unless infected, cause painless swelling around the nasal vestibule and upper lip, and infrequently lead to nasal stuffiness. [3,4] Typically, they appear as a swelling at canine fossa, upper lip, gingivo-labial sulcus, nasal alae and nasal vestibule. [3] Despite the fact that they are soft tissue cysts and are situated extra-osseously, they may sometimes cause bone destruction. [5] The initial diagnosis and treatment is usually made in early stages because the lesion causes cosmetic problems; very rarely it becomes large in dimensions. Commonly seen in adults, it has peak prevalence in the 4 th -5 th decade of life. [6] A greater incidence is seen in females (4:1). It is usually unilateral in occurrence with no predilection in side. [2] However, 11.2% cases have been reported to be bilateral. [7] This paper documents the presentation of nasolabial cyst in a 33 year old woman and discusses considerations related to the diagnosis. CASE REPORT A 33 year old woman was referred to our service for the evaluation of a painless swelling lateral to right ala of the nose that had appeared one month earlier with gradual evolution. Medical history was non-contributory to the present complaint. Extra-oral examination revealed a diffuse swelling lateral to right ala of the nose resulting in elevation of alae and obliteration of the nasolabial fold [Figure 1a]. On palpation, the swelling was soft in consistency, fluctuant and non-tender. On intra-oral examination, swelling distending the right maxillary labial sulcus [Figure 1b] was evident that was soft, fluctuant and non-tender on palpation. A history of extraction with respect to permanent maxillary right first molar and maxillary left first premolar done two years back was the only past dental intervention. The teeth in the vicinity of the swelling showed proximal caries but were tested vital. Intra-oral periapical and panoramic radiographs revealed no obvious bony changes. Maxillary occlusal view revealed slight erosion of right side of the palate due to pressure exerted by the lesion [Figure 2]. Case Report [Downloadedfreefromhttp://www.jofs.inonSaturday,October25,2014,IP:202.67.32.34]||ClickheretodownloadfreeAndroidapplicationforthisjournal Arul, et al.: Nasolabial cyst Journal of Orofacial Sciences Vol. 4 Issue 2 December 2012 134 Aspiration of the swelling yielded a straw-colored fluid [Figure 3]. Based on clinical and radiographic diagnosis, a working diagnosis of nasolabial cyst was made. The clinical differential diagnosis included epidermoid inclusion cyst, salivary gland cyst. Under local anesthesia, using a vestibular incision, the cysts were enucleated and submitted for histopathological examination. Microscopic evaluation revealed a cystic lumen lined predominantly by pseudo-stratified columnar epithelium with varying number of goblet cells [Figure 4a] with part of lining composed of cuboidal epithelium [Figure 4b]. The fibrous cystic wall was relatively acellular, densely collegenous and exceedingly hemorrhagic. The features were consistent with the diagnosis of nasolabial cyst. Post-operative follow up at 8 months showed uneventful healing without evidence of recurrence. DISCUSSION The nasolabial cyst is a developmental, non-odontogenic cyst that most commonly involves the nasal furrow region. [8] According to Allard, the first description of this entity is recorded by Zukerkandl in 1882. [9] It has been given many names such as Klestadts cyst, nasoalveolar cyst, nasal vestibular cyst, mucoid cyst of the nose, and nasal wing cyst. [10] Regarding the pathogenesis, various theories of origin have been proposed. The first suggested that it is a retention cyst arising from inflamed mucus glands. [11,12]
Klestadt first postulated an embryologic origin for these cysts and considered that these lesions must originate from embryonic epithelium, entrapped in the developmental fissures between the lateral nasal and maxillary processes. [13] Since then, many authors have classified this entity based on Klestadts embryologic theory as a fissural cyst. The third theory and the most accepted one raised by Bruggemann is that it arises from the remnants of the lower anterior part of the nasolacrimal duct. [14] The clinical presentation is typical with an asymptomatic spherical swelling beneath the nasal ala causing its elevation and obliterating the nasolabial fold. Lesion distends the mucolabial sulcus intra-orally and can cause discomfort in denture users. [2] Most often patient Figure 1a: Extra-oral photograph showing elevation of right nasal alae and obliteration of the nasolabial fold Figure 1b: Intra-oral photograph depicting the swelling distending right maxillary labial sulcus Figure 2: Maxillary occlusal radiograph revealed slight erosion of right side of the palate Figure 3: Aspiration yielded straw-colored fluid [Downloadedfreefromhttp://www.jofs.inonSaturday,October25,2014,IP:202.67.32.34]||ClickheretodownloadfreeAndroidapplicationforthisjournal Arul, et al.: Nasolabial cyst Journal of Orofacial Sciences Vol. 4 Issue 2 December 2012 135 may seek treatment because of the noticeable deformity, as was in the present case. The diagnosis is essentially clinical. Bi-digital palpation reveals a fluctuating tumefaction between the floor of the nasal vestibule and the gingivolabial sulcus, which helps to confirm the diagnosis. Radiograms do not detect this soft tissue lesion except when it causes significant maxillary bone erosion. [15] Cohen and Hertzanu reported a case of NC with a high growth potential that resulted in the erosion of maxillary alveolus, invaded the supporting structures in the region of incisor teeth and caused their displacement. [16] In the present case, the occlusal radiograph revealed pressure erosion on right side of the palate, but there was no displacement of teeth. The teeth in the lesional area are vital unless affected by the pathosis unrelated to the cyst. [2] In the present case, there was no focus of dental infection in the lesional area and the teeth tested vital. The differential diagnosis for a non-painful vestibular soft-tissue swelling within the anterior maxillary-alar region is not extensive, once conditions other than benign cysts are excluded. However, only nasolabial cyst presents exclusively in this area. Other soft-tissue lesions that can occur in this region include periapical inflammatory lesions (granuloma, cyst or abscess) that have perforated the bone. Vitality testing of the adjacent teeth can help to rule out this possibility. Very rarely, aggressive developmental odontogenic lesions, such as keratocyst, extend through the bone cortex to cause soft-tissue swelling. The long-standing nature of this lesion and the limited bone involvement made this diagnosis improbable. Developmental gingival cyst of the adult has a predilection for the bicuspid or canine region and might have been considered in this case. However, this lesion is usually localized in the gingival or contiguous alveolar mucosa and would not cause the distension of the vestibular mucosa that characterizes nasolabial cyst. Another possible cyst of non-odontogenic origin is the epidermoid or epidermal inclusion cyst. A distinguishing feature of this very rare cyst may be its yellow hue, as opposed to the normal pink or bluish coloration of a nasolabial cyst. Mucous extravasation cyst could also be considered. However, in such cases there is often a history of deflation and inflation as mucus within the lesion is periodically expressed and regenerated. As noted earlier, numerous non-odontogenic benign or malignant neoplasms may present in this area. Of particular significance are salivary gland neoplasms arising from minor salivary glands. [17] Various treatment modalities have been considered for NC including injection of sclerosing agents, marsupalization and surgical enucleation. However, surgical enucleation through sublabial approach is the most accepted treatment modality. Recurrence have never been reported. [2] Malignant transformation is rare and has been documented in only one case. [18] CONCLUSION Nasolabial cyst must be kept in mind in differential diagnosis of nasal vestibule, nasal base, and sublabial area. Although uncommon in occurrence, it is imperative for the clinician to make an accurate diagnosis and provide appropriate treatment. [2] REFERENCES 1. Shear M, Speight PM. 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How to cite this article: Arul AJ, Verma S, Arul AJ, Verma R. Nasolabial cyst: Report of a case. J Orofac Sci 2012;4:133-6. Source of Support: Nil, Confict of Interest: None declared [Downloadedfreefromhttp://www.jofs.inonSaturday,October25,2014,IP:202.67.32.34]||ClickheretodownloadfreeAndroidapplicationforthisjournal