|Year : 2015 | Volume
| Issue : 2 | Page : 292-295
Dentigerous cyst in the maxilla associated with two supernumerary teeth: A rare entity
Sk. Abdul Mahmud, Mousumi Pal, Sanjeet Kumar Das, Ritesh Aich
Department of Oral and Maxillofacial Pathology, Guru Nanak Institute of Dental Sciences and Research, Kolkata, West Bengal, India
|Date of Web Publication||16-Dec-2015|
Sanjeet Kumar Das
B-5/172, Kalyani, Nadia, West Bengal
Source of Support: None, Conflict of Interest: None
A dentigerous cyst is a developmental odontogenic cyst, which apparently develops by accumulation of fluid between the reduced enamel epithelium and the crown of an unerupted tooth. Through various cases that have been reported, it is observed that 95% of this cyst is associated with permanent dentition while only 5-6% is in association with supernumerary teeth. Mesiodens, a supernumerary tooth situated between the maxillary central incisors, has a prevalence of 0.15-1.9%. Here, a dentigerous cyst in association with a pair of impacted mesiodens is reported in a 56-year-old female patient.
Keywords: Dentigerous cyst, mesiodens, supernumerary tooth
|How to cite this article:|
Mahmud SA, Pal M, Das SK, Aich R. Dentigerous cyst in the maxilla associated with two supernumerary teeth: A rare entity. Arch Med Health Sci 2015;3:292-5
|How to cite this URL:|
Mahmud SA, Pal M, Das SK, Aich R. Dentigerous cyst in the maxilla associated with two supernumerary teeth: A rare entity. Arch Med Health Sci [serial online] 2015 [cited 2022 Jan 25];3:292-5. Available from: https://www.amhsjournal.org/text.asp?2015/3/2/292/171928
| Introduction|| |
Dentigerous cysts are the most common developmental odontogenic cysts (10%), arising in relation to impacted, unerupted and rarely in association with impacted supernumerary teeth, caused by expansion of the follicle, being attached to its neck. ,
Usually, they are asymptomatic and often diagnosed accidentally in routine radiographs until they become large enough and destructive to be manifested clinically.
Characteristic histological features include thin epithelial lining backed by loose connective tissue wall simulating ectomesenchyme. ,,,
Treatment of these cysts ranges from marsupialization to enucleation;  and a case report describing an unusual entity of a dentigerous cyst associated with a pair of impacted maxillary mesiodens is presented herewith.
| Case Report|| |
A female patient aged about 56 years reported to our department of Oral and Maxillofacial Pathology, GNIDSR, Kolkata with a chief complaint of mild swelling and discomfort involving the maxillary anterior region for about 1-year, which had aggravated since last 3-4 months. Patient had been aware of the swelling since her premarital days but did not pay any heed to the same. It had been increasing in size very slowly, without any discomfort or pain. She was only aware of the regional tooth mobility for the last 2-3 years that has increased progressively with time.
On intraoral clinical examination, a solitary, well-defined, fluctuant, oval swelling was noted in relation to the maxillary right canine region to the left premolars and extending up to middle of the hard palate, with regional teeth mobility, measuring approximately 8 cm × 5 cm. The overlying mucosa was tensed but without any secondary ulcerative or degenerative changes [Figure 1].
The swelling was fluctuant and well-demarcated from the surroundings. Regional teeth viz. incisors and canines revealed grade I mobility with grade II involving the left maxillary premolars with mild tenderness on percussion. Aspiration yielded a blood tinged brownish fluid. Occlusal radiograph revealed the presence of a well corticated, unilocular radiolucent lesion and a pair of impacted teeth-like structures that appeared to be mesiodens causing displacement of the roots of left maxillary lateral incisors, canine and premolars [Figure 2].
|Figure 2: Maxillary occlusal radiograph showing two impacted supernumerary teeth|
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The patient was primarily diagnosed with dentigerous cyst and was referred to the Department of Oral and Maxillofacial Surgery for the necessary treatment. Enucleation of the cystic lesion along with the removal of the impacted mesiodens were performed [Figure 3] and sent for histopathological evaluation.
The sections stained with H and E revealed the presence of a cystic cavity lined by four to six layers of flattened nonkeratinizing cuboidal epithelial cells backed by connective tissue wall, simulating primitive ectomesenchyme with loosely arranged interlacing collagen fibers and very mild inflammatory cell infiltration, with a flattened epithelial and connective tissue interface. The overall features were as in corroboration with the clinical diagnosis of dentigerous cyst [Figure 4].
|Figure 4: Photomicrograph (high power) showing cystic epithelial lining backed by connective tissue|
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The patient is now on periodic evaluation and doing well.
| Discussion|| |
Dentigerous cyst is one that encloses the crown of an unerupted tooth by expansion of the follicle that is attached to its neck.  This cyst is of odontogenic epithelial origin, occurring due to developmental alterations during odontogenesis. 
It accounts for approximately 20% of all odontogenic cysts being second most common after radicular cyst.  In order of frequency, it is associated with mandibular third molars, maxillary canines, mandibular second premolars and maxillary third molars. , It may also occur around unerupted supernumerary tooth; but rarely with primary dentition.  In our case, we had the association of dentigerous cyst with impacted mesiodens.
Dentigerous cysts are usually asymptomatic in nature, similar to ours; but may develop symptoms due to large size or secondary infection. Bone expansion, pain and swelling are the usual presenting features. 
The pathogenesis of this cyst is based on intrafollicular theory, which postulates the possibility of cyst formation due to fluid accumulation between the layers of outer and inner enamel epithelium after the crown formation. Toller had stated that the likely origin of the dentigerous cyst is the breakdown of proliferating cells of the follicle, after impeded eruption. These breakdown products result in increased osmotic tension and hence cyst formation. 
Radiographically, these cysts appear as unilocular radiolucent areas involving the crown of unerupted teeth, with well-defined sclerotic margins and occasional trabeculations, giving impression of multilocularity.  In our case, we had a large unilocular cystic lesion spanning the anterior maxillary region. However, dentigerous cysts are grossly unilocular lesion and never truly multilocular. 
Supernumerary teeth are normally found in the maxillary anterior region and may cause disorders in dental eruption or alterations in the neighboring teeth. These teeth appear in <1% of the general population. Developmental abnormalities and hyperactivity of the dental plate is the most accepted theory in order to explain their formation. 
Association between dentigerous cysts and supernumerary teeth is an unusual pathology, with the prevalence varying between 1% and 9.9%. According to quite a few published reports, the frequency of impacted supernumerary teeth developing dentigerous cysts varied between 5.5% (Stafne) and 13% (Frietas et al.).  Mesiodens, with an overall prevalence of 0.15-1.9%,  when fail to erupt, is responsible for causing a sequelae of pathological situations like median diastema, dentigerous cyst and delayed eruption. 
Radiographic examination is indicated for diagnosis of mesiodens. The mesiodens are known to have short roots and conical crowns. Their location, number, direction of the crown, influence on adjacent teeth, resorption of roots, etc., should be carefully observed.  Most mesiodens are usually palatal in position to the permanent incisors as in our case. Resorption of root by mesiodens is rarely observed. In our case, we did not see pronounced root resorption, but certain regional teeth displacement had been noted.
The histopathological features may vary greatly depending upon the status of the inflammation. The noninflamed dentigerous cyst is lined by a thin layer of epithelium (which is basically the reduced enamel epithelium), two to four cell layers thick; cells being cuboidal/low columnar, occasionally showing mucous and or squamous metaplasia. As the connective tissue wall is derived from the dental follicle of developing enamel organ, it is a loose connective tissue stroma, with young fibroblasts, with or without the presence of odontogenic epithelial islands. 
Our case depicted features of a noninflamed dentigerous cyst with thin epithelial lining and stroma similar to as described.
Various treatment modalities have been suggested for dentigerous cysts. It may be surgical removal of the cyst avoiding damage to the involved permanent tooth, enucleation of the cyst along with the involved tooth, or use of a marsupialization technique. The nature of the causative tooth influences the type of surgical treatment required for dentigerous cyst. If the cyst is associated with a supernumerary or wisdom tooth, complete enucleation along with tooth extraction is advisable. As in our case, the cyst was associated with a pair of impacted mesiodens; hence this technique had been applied. ,
| Conclusion|| |
Dentigerous cysts associated with impacted mesiodens present an extremely rare entity, presenting varied clinicopathological features. The present case depicted characteristics of a dentigerous cyst in association with a pair of impacted mesiodens, leading to a dome-shaped, fluctuant swelling involving the midpalatine rugae region. Histopathological appearance simulated noninflamed dentigerous cyst. Therefore, proper clinical examination, radiological evaluation and histopathological confirmation, followed by surgical management and subsequent follow-ups are a key to its successful therapy.
| References|| |
Baranwal HC, Verma PK, Dwivedi CD, Srivastava R. Dentigerous cyst associated with an impacted maxillary mesiodens. Eur J Gen 2012;1:50-3.
Barnes L. Surgical Pathology of the Head and Neck. 3 rd
ed., Vol. 2; Informa Healthcare. 2009. p. 1163-6.
Navarro BG, Jané Salas E, Olmo IT, I Muñoz AF, Juarez Escalona I, López-López J. Maxillary dentigerous cyst and supernumerary tooth. Is it a frequent association? Oral Health Dent Manag 2014;13:127-31.
Freitas DQ, Tempest LM, Sicoli E, Lopes-Neto FC. Bilateral dentigerous cysts: Review of the literature and report of an unusual case. Dentomaxillofac Radiol 2006;35:464-8.
Kiso H, Ando P. Dentigerous cyst associated with a supernumerary tooth in the nasal cavity - A case report. J Maxillofac Surg Oral Med Oral Pathol 2015;27:33-7.
Mohan KR, Natarajan B, Mani S, Sahuthullah YA, Kannan AV, Doraiswamy H. An infected dentigerous cyst associated with an impacted permanent maxillary canine, inverted mesiodens and impacted supernumerary teeth. J Pharm Bioallied Sci 2013;5:S135-8.
Motamedi MH, Talesh KT. Management of extensive dentigerous cysts. Br Dent J 2005;198:203-6.
Khambete N, Kumar R, Risbud M, Kale L, Sodhi S. Dentigerous cyst associated with an impacted mesiodens: Report of 2 cases. Imaging Sci Dent 2012;42:255-60.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2 nd
ed. Elsevier. 2009. p. 590-3.
Shear M, Speight P. Cysts of Oral and Maxillofacial Region. 4 th
ed. Blackwell Publishing Ltd. 2007. p. 59-74.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]