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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 4  |  Issue : 2  |  Page : 253-257

Diagnostic enigma of a maxillary unilocular radiolucency with multiple impacted teeth: A case report and review of literature


1 Department of Oral Medicine and Radiology, A. J. Institute of Dental Sciences, Mangalore, Karnataka, India
2 Department of Oral Medicine and Radiology, Bangalore Institute of Dental Sciences, Bengaluru, Karnataka, India

Date of Web Publication20-Dec-2016

Correspondence Address:
Gowri Bhandarkar
Department of Oral Medicine and Radiology, A. J. Institute of Dental Sciences, Kuntikana, Mangalore - 575 004, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2321-4848.196198

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  Abstract 

The unilocular radiolucencies remain the topic of much interest for clinicians for decades. These conditions cannot be diagnosed by clinical appearance alone, but it is based on clinical and radiological features. In spite of the giant strides taken in the field of imaging, there still remains the possibility that an inexperienced clinician may often misdiagnose this entity. It is thus very important for the clinician to have a sound knowledge of various unilocular radiolucencies. Adequate use of diagnostic aids and careful observation will certainly help to arrive at a proper diagnosis and render quality treatment in such cases. Here, we present a case of a rare unilocular lesion in the left anterior maxilla with three impacted teeth.

Keywords: Anterior maxilla, differential diagnosis, multiple impacted teeth, odontogenic cyst, unilocular lesion


How to cite this article:
Pinto MR, Bhandarkar G, Kini R, Naik V, Kashyap RR, Rao PK. Diagnostic enigma of a maxillary unilocular radiolucency with multiple impacted teeth: A case report and review of literature. Arch Med Health Sci 2016;4:253-7

How to cite this URL:
Pinto MR, Bhandarkar G, Kini R, Naik V, Kashyap RR, Rao PK. Diagnostic enigma of a maxillary unilocular radiolucency with multiple impacted teeth: A case report and review of literature. Arch Med Health Sci [serial online] 2016 [cited 2017 Apr 28];4:253-7. Available from: http://www.amhsjournal.org/text.asp?2016/4/2/253/196198


  Introduction Top


Cystic lesions are the most prevalent unilateral swellings of the jaw because of the presence of rests of odontogenic epithelium. A review of the English literature reveals that 0.8–45.9% of the anterior maxillary lesions are odontogenic cysts.[1] Attention to the above malady is drawn, because of their potential complications to supporting dental tissues such as root resorption, local sensory deficits due to nerve involvement and even neoplastic changes in the cystic lining. These lesions could be unilocular or multilocular, usually with well-defined sclerotic borders with or without impacted teeth. They are usually benign, but some can be locally aggressive and destructive.[2]

The patient history and careful consideration of the location of the lesion within the jaw, its borders, internal structure, and effects on surrounding structures generally make it possible to narrow the differential diagnosis. In spite of the narrowed spectrum, many lesions that occur in the jaw tend to mimic and have a similar clinical and radiographic appearance. Therefore, unilocular osteolytic radiolucencies still remain a diagnostic dilemma for clinicians.[3]

Here, we present a case of a rare unilocular lesion of left maxillary anterior region involving three impacted teeth. Furthermore, we submit a systematic literature review with a clinical and radiological perspective concerning cases of unilocular osteolytic lesions of the anterior maxilla in association with multiple impacted teeth, emphasizing on differential diagnosis.


  Case Report Top


A male patient aged 27–year-old reported to our dental outpatient department, with chief complaint of swelling in relation to the upper left front tooth region, which he noticed since 15 days. An associated history of trauma at the age of seven was elicited, following which there was a failure of regional permanent teeth. There were no symptoms such as pain and fever. On extraoral examination, a diffuse unilateral swelling was noticed in the left upper anterior tooth region, measuring about 2.5 cm × 2 cm in diameter, extending mediolaterally from the filtrum to the nasolabial fold obliterating it, superio-inferiorly from the 1 cm above ala of nose to the vermillion border of the upper lip, with no visible color changes [Figure 1]. On palpation, there was no local rise in temperature; swelling was nontender and hard in consistency.
Figure 1: Extraoral swelling showing obliteration of nasolabial fold.

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Intraorally, a well-defined solitary oval-shaped swelling measuring about 2.5 cm × 2 cm in diameter was seen on the gingiva of the upper left tooth region extending to the labial vestibule and obliterating it with labial cortical plate expansion. Overlying mucosa was intact without color changes [Figure 2]. Swelling was nontender, compressible suggestive of cystic consistency. Maxillary left incisors and canine were clinically missing. Deciduous lateral incisor and canine were retained, attrited, nontender, and discolored with Grade II mobility.
Figure 2: Intraoral swelling seen on upper left labial mucosa showing vestibular obliteration.

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Based on the history and clinical presentation, a provisional diagnosis of inflammatory odontogenic cysts such as radicular cyst in relation to central incisor, lateral incisor, and canine was considered. Dentigerous cyst and adenomatoid odontogenic tumor (AOT) were thought of as differential diagnosis.

On fine-needle aspiration, a straw-colored fluid was aspirated which is further suggestive of an odontogenic cyst. Radiological investigations with an occlusal view revealed a well-defined unilocular radiolucency with sclerotic borders on left anterior region, measuring about 3 cm × 1 cm in diameter, enclosing the crowns of impacted permanent lateral incisor and canine present in the vicinity. Displacement of impacted permanent central incisor was also noted [Figure 3]. Further on orthopantamographic view, a superiorly displaced tooth like radiopacity suggestive of impacted left central incisor was seen at the superior border of the existent radiolucency [Figure 4]. With the above facts, dentigerous cyst, AOT and radicular cyst were considered as radiological differential diagnoses.
Figure 3: Occlusal view showing unilocular radiolucency involving necks of two permanent teeth.

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Figure 4: Orthopantamograph showing a well-.defined unilocular radiolucency involving the necks of permanent left lateral incisor and canine with left central incisor lying at superior border.

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In our case, complete enucleation of cyst along with the extraction of left deciduous and permanent incisors and canine had been planned followed by bone grafting and prosthodontic rehabilitation of missing teeth.


  Discussion Top


Odontogenic cysts account for 0.8–45.9% of the lesions diagnosed in the oral cavity.[1] They deserve attention because of their potential complications including damage to supporting dental tissues/jaw bone, like root resorption, local sensory deficits due to nerve involvement and even neoplastic changes in the cystic lining.[4]

Despite the development of various cross-sectional imaging modalities, the radiograph still remains the first-line of simple investigation. Any jaw lesion should be evaluated taking into consideration the following radiological features.[3],[5]

  • Density, margin, and locularity of lesions
  • Anatomical location, and relation to dentition
  • Cortical integrity, periosteal, and soft tissue reaction
  • Effect on surrounding structures.


When the clinician is confronted with unilocular radiolucencies, a list of possible diagnoses should be formulated and arranged in order of probability, with the most probable lesion heading the list. It is thus very important for the clinician to have a sound knowledge of various unilocular radiolucencies. In our case, based on the age, site, type of lesion various differential diagnosis considered were, radicular cyst, dentigerous cyst, unicystic ameloblastoma, AOT, keratocystic odontogenic tumor (KCOT), ameloblastic fibroma, calcifying odontogenic cyst, and calcifying epithelial odontogenic tumor (CEOT).

Radicular cyst

The radicular cyst is most common inflammatory odontogenic cyst of the jaw. Greater incidence is seen in males in the third to sixth decades of life. Around 60% are found in the maxilla, especially around incisors and canines.[6] A swelling is seen in the case of larger cyst which on palpation, may feel bony hard if the cortex is intact.

Radiographically, epicenter is located approximately at the apex of a nonvital tooth. Well-defined cortication surrounds the periphery of radiolucency. Due to continuous increase in size, it often causes displacement and resorption of the roots of adjacent teeth. Association with a nonvital deciduous tooth is a rare possibility. The cyst present at the apex of the primary tooth may surround the permanent tooth bud, giving false impression of a dentigerous cyst. In these cases, the clinician should look for deep caries or extensive restorations in a primary tooth that would indicate a radicular cyst.[6] As radicular cyst rarely develops associated with primary teeth, radicular cyst was ruled out in our case.

Unicystic ameloblastoma

Unicystic ameloblastoma is a variant of ameloblastoma which mimic an odontogenic cyst, both clinically and radiographically. It typically presents at a younger age group approximately 21.8 years with no sex predilection. The pretentious site is mandibular third molar region. Predominantly, asymptomatic lesion which remains undetected until radiolucency is seen on the routine radiograph.[4] As the lesion slowly enlarges, a slight, nontender hard swelling becomes apparent on clinical examination. There is often expansion, thinning and destruction of cortical plates.[5]

Radiographically, it appears as a well-defined unilocular radiolucency when it is associated with an unerupted tooth it is arduous to distinguish from the dentigerous cyst. In view of the fact that expansion of buccal cortical plate only and no resorption of adjacent teeth unicystic ameloblastoma was excluded.

Adenomatoid odontogenic tumor

AOT is a relatively uncommon distinct odontogenic tumor, presents in the second decade of life in women, with an average age of 16 years. 75% of them occur in maxilla at incisor-canine-premolar region. A slow growing painless swelling causes asymmetry in the affected region. There is often a missing tooth in the vicinity.[6]

The usual radiographic appearance is well-defined radiolucency with corticated or sclerotic border. Calcifications within tumor produce faint radio-opacities in about two-thirds of cases. Larger radiolucencies displace the adjacent teeth, but root resorption is rare. This lesion may inhibit eruption of an involved tooth. These tumors surround the crown and root of the involved tooth. Evidence of a radiopaque internal structure should be sought in these lesions.[6] As there was no internal radiopacity in the unilocular lesion and radiolucency was involving only the neck of the teeth, AOT was excluded.

Keratocystic odontogenic tumor

KCOT accounts for about one-tenth of all cysts in the jaw. They occur in a wide age range, but most develop during the second and third decades, with a slight male predominance. The most common location is the posterior body and ramus of the mandible. The cysts sometimes form around an unerupted tooth. They are usually asymptomatic, although mild swelling may occur. They can displace and resorb teeth.[4] An important characteristic is its propensity to grow along the internal aspect of the jaws, causing minimal expansion.

Radiographically, well-defined round or oval-shaped radiolucency with scalloped outline may be seen. The internal structure most commonly is radiolucent. In some cases, curved internal septa may be present, giving the lesion a multilocular appearance.[6] In our case, on aspiration straw colored fluid was aspirated as against thick, yellow cheesy material containing keratin seen in KCOT and also based on the fact of minimal expansion this tumor was excluded.

Ameloblastic fibroma

It is a rare benign tumor which occurs between 5 and 20 years of age, during the period of tooth formation, with no sex predilection. It arises frequently in the premolar-molar area of the mandible. They usually produce a painless, slow growing expansion and displacement of the involved teeth. It may be associated with a missing tooth.[5] Radiographically, appears as a well-defined, usually unilocular or multilocular radiolucency often with corticated border as that of a cyst. The associated tooth or teeth may be inhibited from normal eruption or may be displaced in an apical direction.[6]

In our case, the patient was 27-year-old and the absence of radiopacities radiographically lead to exclusion of this lesion.

Calcifying odontogenic cyst (Gorlin cyst)

The calcifying odontogenic cyst mostly presents in the second or third decade of life. Usually, maxilla and mandible are affected equally, particularly in the area of the incisors and canines. Radiographically, appears as a well-defined unilocular or multilocular radiolucency. In about a third of cases, radiopaque structures are present within the lesion. These cysts surround the crown and root of the involved tooth.[2] In our case, due to the absence of radiopacities within the radiolucency Gorlin cyst was excluded.

Calcifying epithelial odontogenic tumor

The CEOT is a rare, benign epithelial neoplasm, more common in men. It occurs over a wide age range from 8 to 92 years with an average age of about 42 years. It has a predilection for mandible in the premolar-molar area, with a 52% association with a unerupted tooth or impacted tooth. The chief sign is cortical expansion without pain. Palpation of the swelling reveals a hard tumor that may be quite well-defined or diffuse.[6]

Radiographs reveal a unilocular or multilocular cystic lesion with numerous scattered, radiopaque foci of varying size and density. Perhaps, may be associated with crown of an unerupted tooth, normally displacing it from the path of eruption. Associated expansion of the jaw with the maintenance of a cortical boundary may also occur. Early lesions may mimic dentigerous cysts or even ameloblastomas.[5] Owing to the age of occurrence, (>42 years) and absence of internal radiopacities, CEOT was a distant possibility.

Dentigerous cyst

Dentigerous cyst is the second most common developmental odontogenic cyst which encloses the crowns of unerupted, impacted, embedded permanent teeth, supernumerary teeth, or odontomas at the level of cementoenamel junction (CEJ).[7] Based on pathogenesis, it can be of two types:First is developmental in origin and occurs in impacted mature teeth. Second is inflammatory in origin and occurs in immature teeth as a result of inflammation from a nonvital deciduous tooth follicle.[8]

It begins when fluid accumulates in the layers of reduced enamel epithelium or between the epithelium and the crown of the unerupted tooth. They are more common in male subjects occurring most often in second and third decades of life with occurrence rate of 70% and 30% in mandible and maxilla, respectively.[8] Dentigerous cyst involving multiple teeth is uncommon.

The clinical examination reveals a missing tooth or teeth and possibly a painless hard swelling, occasionally resulting in facial asymmetry. This is a slow-growing cyst and often expands the outer cortical boundary of the involved jaw and seldom erodes it. An important diagnostic point is that this cyst attaches at the CEJ.[6],[7]

Radiographically, a well-defined unilocular radiolucency of varying sizes with corticating margins in association with the crown of an unerupted tooth is evident.[7] The internal aspect is completely radiolucent except for the crown of the involved tooth.[6] It has a propensity to displace and resorb adjacent teeth and also the associated tooth in an apical direction.

In view of the fact that expansion of buccal cortical plate only and no resorption of adjacent teeth it was considered as a low growing cyst. Hence, cyst may not have been noticed by the patient and was noticed by him only in the recent days. In our case, unilocular radiolucency was situated in the anterior maxilla associated with unerupted incisors and canine with attachment at the CEJ favoring dentigerous cyst.

Recent concepts in pathogenesis of dentigerous cyst

The first theory suggests dentigerous cyst being developmental in origin, occurs mostly in mature teeth as a result of impaction.[8] It has been suggested that the pressure exerted by a potentially erupting tooth on an impacted follicle obstructs the venous outflow and thereby inducing rapid transudation of serum across the capillary walls. The increased hydrostatic pressure of this pooling fluid separates the follicle from the crown either between the reduced enamel epithelium and the enamel or alternatively between individual layers of the reduced enamel epithelium.[7],[8] Toller suggests that the likely origin of cyst is breakdown of proliferating cells of the follicle after impeded eruption. These breakdown products result in increased osmotic tension and hence cyst formation.[8]

The second theory advocates that radicular cyst originating from the primary predecessor impedes the eruption path of the immature permanent tooth bud.[9] This is the least accepted hypothesis because radicular cysts rarely develop associated with primary teeth.[10] According to the third theory, the periapical infection of the primary predecessor caused inflammation of the periapical tissues which would reach and stimulate the developing permanent tooth germ, thus providing accumulation of fluid and hence dentigerous cyst.[8],[10]

Two possible theories have been speculated to explain the multiple teeth in the single dentigerous cyst, one is a fusion between two adjacent dentigerous cyst linings, and the other is a fusion between the lining of one preexisting cyst and the lining of reduced enamel epithelium surrounding the adjacent tooth. Another possibility as suggested by Agrawal et al. is that the tooth pushed ectopically further away from the alveolus (due to the expansion of the cyst) tends to erupt in the cyst lining in the same fashion as it erupts in the oral mucosa.[8]

As regarding the impaction and displacement of maxillary permanent central incisor which is quite rare, could be because of alteration in the path of eruption of the tooth caused by the early onset of the unilocular lesion surrounding permanent maxillary canine and lateral incisor.


  Conclusion Top


The present case highlights the fact that apart from the involvement of maxillary canine, the cyst was also found to involve the lateral incisor, which is uncommon. Due to the presence of nonvital deciduous teeth and unerupted permanent counterparts, the etiology was considered of inflammatory origin.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Açikgöz A, Uzun-Bulut E, Özden B, Gündüz K. Prevalence and distribution of odontogenic and nonodontogenic cysts in a Turkish population. Med Oral Patol Oral Cir Bucal 2012;17:e108-15.  Back to cited text no. 1
    
2.
George G, Padiyath S. Unicystic jaw lesions: A radiographic guideline. J Indian Acad Oral Med Radiol 2010;22:s31-6.  Back to cited text no. 2
  Medknow Journal  
3.
Neyaz Z, Gadodia A, Gamanagatti S, Mukhopadhyay S. Radiographical approach to jaw lesions. Singapore Med J 2008;49:165-76.  Back to cited text no. 3
    
4.
Gill Y, Scully C. Orofacial odontogenic infections: Review of microbiology and current treatment. Oral Surg Oral Med Oral Pathol 1990;70:155-8.  Back to cited text no. 4
    
5.
Wood NK, Goaz PW. Differential diagnosis of oral and maxillofacial lesions. 5th ed. Missouri: Elsevier Mosby; 2007. p. 279-95.  Back to cited text no. 5
    
6.
White SC, Pharaoh MJ. Oral Radiology. Principles and Interpretation. 4th ed. St. Louis: Mosby; 2000. p. 359-61, 386-400.  Back to cited text no. 6
    
7.
Ganesh P, Anehosur V, Joshi A, Gopalkrishnan K. Dentigerous cyst of maxilla involving multiple impacted teeth: A rare case report. Int J Oral Maxillofac Pathol 2012;3:45-9.  Back to cited text no. 7
    
8.
Agrawal M, Raghavendra PD, Singh B, Agrawal N. Multiple teeth in a single dentigerous cyst follicle: A perplexity. Ann Maxillofac Surg 2011;1:187-9.  Back to cited text no. 8
[PUBMED]  Medknow Journal  
9.
Ertas U, Yavuz MS. Interesting eruption of 4 teeth associated with a large dentigerous cyst in mandible by only marsupialization. J Oral Maxillofac Surg 2003;61:728-30.  Back to cited text no. 9
    
10.
Aggarwal P, Sohal BS, Uppal KS. Dentigerous cyst of mandible. Int J Head Neck Surg 2013;4:95-7.  Back to cited text no. 10
    


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