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| Year : 2007 | Volume
: 18
| Issue : 1 | Page : 23-26 |
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| Klestadt's cyst: A rarity |
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Karthikeya Patil, VG Mahima, A Divya
Department of Oral Medicine and Radiology, J.S.S. Dental College and Hospital, S. S. Nagar, Mysore - 570015, India
Click here for correspondence address and email
| Date of Submission | 16-Feb-2006 |
| Date of Decision | 17-Oct-2006 |
| Date of Acceptance | 31-Oct-2006 |
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 Abstract | | |
Klestadt's cyst, more commonly known as the nasolabial cyst is an uncommon, non-odontogenic, soft tissue cyst characterized by its extra osseous location in the nasal alar region.[2] Following its description first by Zukuerkandl in 1882,[3] only 267 cases have been found in English literature. A case of nasolabial cyst in a 30-year-old female patient is presented. Keywords: Computed tomography, contrast media, cyst, Klestadt′s cyst, nasolabial, non-odontogenic, soft tissue cyst
How to cite this article:
Patil K, Mahima V G, Divya A. Klestadt's cyst: A rarity. Indian J Dent Res 2007;18:23-6 |
| Introduction | |  |
The head and neck region comprises one of the most common sites for the occurrence of cysts.[1] They can occur within the jaws or the soft tissues in the oro-facial region. They can be developmental or inflammatory in origin. Klestadt's cyst (nasolabial cyst) is one such developmental, non-odontogenic soft tissue cyst occurring in the nasolabial region. It accounts for only 0.7% cysts of the jaws.
| Case Report | | |
A 30-year-old female patient complained of swelling of 2 months duration on the left side of the face adjacent to the nose. The swelling had been gradually increasing in size. Patient was asymptomatic, except for the swelling. Patient's medical, dental, family and personal histories were non- contributory. General examination revealed a moderately built and nourished patient with satisfactory vital signs. The face appeared asymmetric due to the swelling in the left paranasal region [Figure - 1].
On extraoral examination, a diffuse swelling was evident in the left paranasal region, extending medio-laterally from the left ala of the nose to 1cm lateral to it and supero-inferiorly from the infraorbital margin to 1cm superior to the corner of the mouth. The skin overlying the swelling did not show any secondary changes. The left nasal ala appeared elevated and left nasolabial fold was obliterated [Figure - 1]. The swelling was non-tender, soft in consistency, fluctuant and mobile. Intranasally, there was obliteration of the left nasal vestibule on lateral aspect.
Intraoral examination revealed a swelling that distended the left maxillary labial sulcus. The mucosa overlying the swelling exhibited a bluish tint [Figure - 2]. The swelling was non-tender, soft and fluctuant. The teeth in the vicinity of the swelling did not show any discoloration or structural change and tested positive with an electric pulp tester. Aspiration of the swelling yielded blood tinged straw-colored fluid.
Based on the history and clinical findings, a provisional diagnosis of nasolabial cyst was given. The clinical differential diagnosis included epidermoid cyst, dentigerous cyst associated with impacted supernumerary tooth, and odontogenic keratocyst in relation to 22, 23. The patient was subjected to radiographic and histopathological investigations.
Intraoral periapical radiograph and orthopantomograph did not reveal any bony changes. Occlusal view revealed erosion on the left side of the palate due to pressure exerted from the lesion [Figure - 3].
Ultrasound scanning revealed a well-defined anechoic area in the left naso-maxillary region measuring around 2.6 x1.8 sq. cm, with clear contents and sediments at the bottom suggestive of a soft tissue cyst in the left naso-maxillary region.
Around 2 ml of cystic fluid was aspirated. An iodine-based aqueous contrast medium was injected into the cyst and series of radiographs were made. Orthopantamograph showed the contrast media defining the cyst apical to 21, 22, and 23. Posterio-anterior view of skull showed the contrast media defining the cyst in the soft tissues of the left naso-maxillary region and lateral view of skull confirmed the extra-osseous nature of the cyst [Figure - 4]. Axial and coronal view of computed tomograph, apart from emphasizing the extra-osseous nature of the cyst also revealed the deviation of nasal septum to right side and depression of the maxillary bone in the anterior region on left side suggestive of a benign lesion in the left naso-maxillary region [Figure - 5].
Through an intraoral approach, the mucosa was separated easily from the cyst wall with blunt dissection and cyst was enucleated off with an intact nasal mucosa.
Microscopically, the section showed pseudostratified columnar epithelium and stratified squamous epithelium lining the cystic lumen with fibrous connective tissue and chronic inflammatory cells, suggestive of nasolabial cyst [Figure - 6]. Based on the clinical, radiographic and histopathological findings a final diagnosis of nasolabial cyst was arrived at. The postoperative course was uncomplicated and there was no recurrence of the lesion at six months of follow-up.
| Discussion | | |
Klestadt's cyst is named after the author who postulated its origin. The cyst is synonymous with nasolabial cyst, nasoalveolar cyst, nasal vestibule cyst, nasal wing cyst and mucoid cyst of the nose.[4]
Nasolabial cyst (NC) is a rare lesion accounting for 0.7% of all the cysts in the maxillo-facial region and 2.5% of all non-odontogenic cysts.[3],[4],[5] However, many authors believe its prevalence to be higher than that quoted in literature because of misdiagnosis.[3] It is more common in African-Americans.[6] Commonly seen in adults, it has peak prevalence in the fourth and fifth decade of life.[7] A greater incidence is seen in females (4:1). It is usually unilateral in occurrence with no predilection for side.[3] However, 11.2% of cases have been reported to be bilateral.[8]
There are two etiological theories proposed.[5],[6],[7] Klestadt in 1913 suggested that NC arises from entrapped embryonic epithelium at the junction of the medial nasal, lateral nasal and maxillary processes, hence the term 'fissural cyst'.[8] This theory was discarded owing to the purely soft tissue involvement of the cyst and by the lack of evidence for embryonic epithelial entrapment in this region.[5],[6],[8] Bruggeman in 1920, suggested the origin of NC from the remnant of embryonic nasolacrimal duct. This is supported by the fact that nasolacrimal duct is lined by pseudostratified columnar epithelium, the epithelium found in the cystic cavity of nasolabial cyst and also the location of the lesion being subjacent to the wing of the nose in line with naso-optic fissure.[5],[8]
The clinical presentation of NC is typical with an asymptomatic spherical swelling beneath the nasal ala causing its elevation and obliterating the nasolabial fold.[5],[7] The swelling may encroach upon the lateral wall of the nasal vestibule to a varying degree causing nasal obstruction.[8] Lesion distends the mucolabial sulcus intraorally and can cause discomfort in denture users.[3],[7]
NC does not cause any displacement of the teeth. However, Cohen and Hertzanu in 1985 have 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 displacement of these teeth.[4] In the present case the occlusal radiograph revealed pressure erosion on the left side of the palate. There were no signs of displacement of teeth.
The lesion is usually asymptomatic unless it is secondarily infected which causes pain.[5],[7] Referred pain to the maxillary anterior teeth can occur when the lesion increases in size.[7],[8] Most often patient may seek treatment because of the noticeable deformity,[4] as was in the present case. The cyst is soft, fluctuant and is best palpated bimanually with one finger on the floor of the nose and one finger in the labial sulcus.[4],[6]
The teeth in the area of lesion are vital unless affected by a pathosis unrelated to the cyst.[5] Sometimes the cyst may rupture spontaneously and drain into the oral cavity or nose.[7] In the present case, there was no dental focus of infection in the area of lesion and the teeth were vital.
It is important to clinically differentiate nasolabial cyst from other lesions presenting at this site. Periapical pathologies like granuloma, cyst or abscess, when they are infected and perforate the bone simulate the NC. In such cases, vitality test helps in differentiating NC from the above pathosis. The present case presented a positive response to vitality test and thus the existence of periapical pathology was excluded. Epidermoid cyst has a yellowish tint of the overlying mucosa unlike NC, which has bluish tint of the overlying mucosa. Mucocele will have a history of trauma in the nasolabial region, and will exhibit fluctuation in the size of the swelling. Other lesions that could be included in the differential diagnosis are lipoma,[9]salivary gland tumors and odontogenic keratocyst.[9],[10] However, the rarity of occurrence of these lesions at this site should be considered.
Routine intraoral periapical radiographs assist in exclusion of other cysts of embryologic or odontogenic origin that may occur in the same area.[5] NC can cause depression of the underlying labial surface of the maxillary bone due to pressure, a finding detectable by a tangential view.[5] This pressure resorption of the maxilla was apparent on the computed tomography (CT) in the present case too. Rare cases of radicular resorption due to this cyst have been reported.[3] NC may also cause distortion of the curve of the 'bracket - shaped' line produced by the inferior margin of the anterior bony aperture of the nose.[5]
Another technique that helps in the diagnosis of the NC is aspiration of the cyst fluid and replacement of the fluid with a contrast medium.[4],[5] The lesion is then viewed with tangential, posteroanterior or occlusal radiographs. The procedure aids in confirming the extraosseous nature of the NC along with defining its size, site and boundaries. The procedure carries a risk of infection, which must be weighed against the advantage of carrying out this technique.[5],[11]
In the present case, the cyst was asymptomatic and hence it was possible to perform this procedure. The procedure helped us in delineating the extensions of the lesion and confirming the extraosseous nature of the lesion. CT and magnetic resonance imaging help in providing soft tissue images, in delineating the extensions of the lesion and studying the effects of the cyst on surrounding structures.[4],[6] In the present case, CT scan was performed especially to identify the effect of cyst on the surrounding bone. It revealed a depression in the anterior maxillary bone, which may be attributed to the pressure effect from the chronic nature of the lesion.
Diagnosis of NC is established by correlation of the clinical findings with the histological examination.[5],[12] Cystic cavity is commonly lined by pseudostratified columnar epithelium. Areas of stratified squamous cells may also be present and numerous goblet cells may be seen within the epithelium. The cyst wall is either of dense mature fibrous nature or may be made up of loose connective tissue. Hemorrhage and chronic inflammatory cell infiltration are often present that correlate with the clinical symptom of pain that patients experience.[4],[5],[6] In the present case, the cystic cavity presented both pseudostratified columnar and stratified squamous epithelial lining. The connective tissue was fibrous and contained chronic inflammatory cells mainly lymphocytes.
Various treatment modalities for the NC have been considered including injection of sclerosing agents, marsupalization and surgical enucleation.[3] However, surgical enucleation through sublabial approach is the most accepted mode of treatment.[3],[6] Recurrence has never been reported.[3],[4],[6] Malignant transformation is rare and has been documented in only one case.[6]
| Conclusion | | |
NC is a benign cyst and very few cases have been documented. Although uncommon in occurrence, it is imperative for the clinician to make an accurate diagnosis and provide appropriate treatment. In the present case the diagnosis was based on the correlation of clinical, radiographical and histopathological features.
As NC is a soft tissue cyst, plain radiography does not depict any characteristic findings. Contrast radiography plays an important role in defining the borders and extent of the cyst, which further helps in diagnosis and treatment planning. It is prudent to make use of this procedure whenever required. However complications such as secondary infection should be kept in mind.
| Acknowledgement | | |
The authors thank Dr. K. Praveen. MD., Kannan Diagnostic Center, Mysore. Dr. SaiKrishna. MDS., Department of Oral and Maxillofacial Surgery, J.S.S Dental College and Hospital, Mysore. Dr. Sudheendra. MDS., Department of Oral and Maxillofacial Pathology, J.S.S Dental College and Hospital, Mysore.
| References | | |
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| 3. | Pereira Filho VA, Da Silva AC, Moraes MD, Moreira RWF, Villalba H. Nasolabial cyst: Case report. Braz. Dent. J 2002;13:212-14. |
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| 7. | Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. Ed 2nd. Philadelphia. W.B. Saunders company.25-26. |
| 8. | Wesley RK, Scannel T, Nathan LE. Nasolabial cyst: Presentation of a case with a review of the literature. J Oral Maxillofacial Surg 1984;42:188-92. |
| 9. | Wood NK, Goaz Pw. Differential diagnosis of oral lesions. Ed 5th Mosby 2005. |
| 10. | Lucas RB. Pathology of tumours of the oral tissues. Ed 4th. Edinberg: Churchill Livingstone 1984.p.362. |
| 11. | Campbell RL, Burkes EJ. Nasolabial cyst: Report of case. J Am Dent Assoc 1975;91:1210-13. |
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Correspondence Address:
Karthikeya Patil
Department of Oral Medicine and Radiology, J.S.S. Dental College and Hospital, S. S. Nagar, Mysore - 570015
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0970-9290.30918
Figures
[Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5], [Figure - 6] |
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