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| CASE REPORT |
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| Year : 2009 | Volume
: 21
| Issue : 3 | Page : 119-121 |
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Botryoid odontogenic cyst
Vandana Arya, M Jonathan Daniel
Department of Oral Medicine and Radiology, Mahatma Gandhi Postgraduate Institute of Dental Sciences, Puducherry, India
| Date of Web Publication | 7-Jan-2010 |
Correspondence Address:
Vandana Arya
Department of Oral Medicine and Radiology, Mahatma Gandhi Postgraduate Institute of Dental Sciences, Puducherry
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0972-1363.58752
 Abstract | | |
Botryoid odontogenic cyst is an unusual type of cyst. It was first reported by Weathers and Waldron in 1973. It is simply a polycystic variant of the lateral periodontal cyst developing through cystic transformation of multiple islands of dental lamina rests. A case of botryoid odontogenic cyst in a 35-year -old female patient with its clinical, radiological and histopathological presentation is reported. Keywords: Botryoid odontogenic cyst, poly cystic variant, recurrence
How to cite this article:
Arya V, Daniel M J. Botryoid odontogenic cyst. J Indian Acad Oral Med Radiol 2009;21:119-21 |
| Introduction | |  |
The botryoid odontogenic cyst is very rare in occurrence; only 67 cases have been reported from 1973 to the last case published in the year 2005. [1]
The botryoid odontogenic cyst is named for its characteristic multilocular appearance which resembles a cluster of grapes. The botryoid odontogenic cyst is considered to be a multiloculated variant of the lateral periodontal cyst. [2],[3]
Most often, it presents as an asymptomatic multilocular radiolucency lateral to the root of a vital mandibular cuspid or premolar tooth and rarely in the anterior maxillary region. It is a variant of lateral periodontal cyst. The two cysts are identical in epithelial lining, clinical features, age predilection and site of occurrence.
Treatment is surgical enucleation. It has increased risk of recurrence or persistence. Patient should be followed periodically.
| Case Report | | |
A 35-year-old female patient presented with a swelling in the anterior palatal region [Figure 1]. Intra-oral examination revealed 3 x 3 cm swelling extending from 12 to 14 up to the midpalatal raphae [Figure 2]. The overlying mucosa was the same as that of the adjacent region, and the surface was smooth. There was mild fullness of the buccal vestibule in relation to 11, 12, 13, 14 regions, due to the swelling [Figure 3]. Thermal vitality test revealed delayed response in 13 and 14. Aspiration was non productive.
On the basis of clinical examination, the provisional diagnosis of benign odontogenic tumor probably ameloblastoma was given.
IOPA radiograph showed honey-comb type of multilocular radiolucency in relation to 12, 13, 14, with irregular, jagged borders [Figure 4].
Maxillary occlusal radiograph confirmed IOPAR findings and showed no significant buccal cortical expansion in the region of the lesion [Figure 5].
Panoramic radiographs revealed well -defined multilocular radiolucency in the region of 11, 12, 13, 14 teeth with a sclerotic border [Figure 6]. There was a partial loss of lamina dura in 12 and 13. There was no evidence of root resorption.
Subsequently, differential diagnosis of salivary gland tumor, ododntogenic cyst or lateral periodontal cyst was considered.
Histopathological examination revealed well -defined lining epithelium with most areas of flat epithelium, but showing plaque -like thickening in some areaws. There was polarized arrangement of basal cells [Figure 7]. Apart from this, multiple small cysts were noticed in the capsule. The underlying connective tissue capsule was relatively free from inflammation.
The features were suggestive of lateral periodontal cyst probably botryoid type.
| Discussion | | |
The botryoid odontogenic cyst is a developmental cyst of odontogenic epithelial origin considered as a rare multilocular variety of lateral periodontal cyst (WHO 1992). [4]
It was first reported by Weathers and Waldron (1973). [5] They reported two cases of multilocular cystic lesions of the jaws for which they proposed the term 'botryoid odontogenic cyst' because the gross specimen resembled a cluster of grapes. [6]
Usually, the cystic lesion site is in the periodontal space of vital teeth which is also observed in the present case. This is found in the premolar region of mandible, as well as in the frontal region of the maxilla of patients aged between 60 and 70 years. [7] In the present case, site of the lesion is in the anterior maxillary region which has been also quoted by Chbicheb S, Bennani A et al. (2008). [4] Kaugars (1986) [6] reported three cases, one which occurred in the midline of the mandible, one between the mandibular premolars and one in an edentulous mandible. All the three patients were in their fifties.
According to various studies, the age of occurrence of botryoid odontogenic cyst is between 20 and 70 years. The age of the female patient in the present case is 35 years which is similar to the case presented by O. Ucok, et al. [8]
Green and Johnson (1988) reported 10 cases. [6] The ages of the patients ranged from 26 to 66 years with a mean age of 46 years; eight of the 10 lesions were in mandible, predominantly the anterior region; eight showed unilocular radiolucency and two were multilocular, ranging in size from 0.4 to 4.5 cm. Three cases showed recurrences, 8, 10, and 11 years, respectively, after previous surgery, and the authors reported Kaugar's concern that patients must be followed up.
Histopathologically, the cyst is marked by multilocular cysts lined by a thin, nonkeratinized epithelium. In our case, the histopathological features confirmed the diagnosis. There were multiple small cysts in the capsule and also there was plaque -like nodular thickening of cyst lining. Sometimes, clusters of glycogen rich epiethelial cells may be noted in nodular thickening of the cyst lining.
Radiographically, it appears as a multilocular radiolucent area in apposition to the lateral surface of a tooth root. The lesion is usually small in diameter and may or may not be well circumscribed. In most cases, the border is definitive and is even surrounded sometimes by a thin layer of sclerotic bone. In the present case, the findings revealed a well-defined multilocular radiolucency in relation to the involved teeth, with a definitive sclerotic border and showing no evidence of root resorption.
It should be treated by conservative surgical excision. [9] Attempts should be made to avoid sacrificing the associated tooth. As it has an increased risk of recurrence [10] or persistence, it should be followed periodically. Recurrence rate of cases with follow-up data is 32.4% (Mιndez P et al.). [1]
| Conclusion | | |
Although botryoid odontogenic cyst is a rare condition, its diagnosis is especially important because of the similarity in appearance between this cyst and other more serious lesion such as an early ameloblastoma. It should be followed periodically for recurrence. Size and multilocular patterns could be the main factors associated with recurrence in botryoid odontogenic cyst.
| References | | |
| 1. | Mιndez P, Junquera L, Gallego L, Baladrσn J. Botryoid odontogenic cyst: Clinical and pathologic analysis in relation to recurrence. Med Oral Patol Oral Cir Bucal. 2007 Dec 1;12(8):E 594-8.  |
| 2. | Gurol M, Burkes EJ Jr, Jacoway J. Botryoid odontogenic cyst: Analysis of 33 cases. J Periodontol. 1995 Dec;66(12):10. |
| 3. | Carter LC, Carney YL, Perez-Pudlewski D. Lateral periodontal cyst. Multifactorial analysis of a previously unreported series. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1996 Feb;81(2):210-6. |
| 4. | Chbicheb S, Bennani A, Taleb B, Wady WE. Botryoid Odontogenic Cyst; A Case Report. Rev Stomatol Chir Maxillofac. 2008 Apr; 109(2):114-6. Epub 2008 Mar 14. |
| 5. | Shafer's Textbook of Oral Pathology Fifth Edition. |
| 6. | Shear cysts of the oral regions 3 rd edition. |
| 7. | Weibrich G, Kleis WK, Otto M, Gnoth SH, Burwinkel M, Kriegsmann J, Kuffner HD, Wagner W. Cytokeratin expression in botryoid odontogenic cyst. A rare differential keratocyst and ameloblastoma diagnosis. Mund Kiefer Gesichtschir. 2000 Sep 4(5):309-14. |
| 8. | άηok Φ, Yaman Z, Gόnhan Φ, Doπan CN and Baykul T. Botryoid odontogenic cyst: report of extensive epithelial proliferation. International Association of Oral and Maxillofacial Surgeons 2005, 26 January. |
| 9. | Burket's Oral Medcine 11 th edition. |
| 10. | Greer RO Jr, Johnson M. Clinicopathologic analysis of ten cases with three recurrences. J Oral Maxillofac Surg. 1988 Jul; 46(7):574-9. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]
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