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 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 3  |  Issue : 2  |  Page : 198-200

Unicystic ameloblastoma associated with impacted supernumerary premolar: An unusual case report


1 Department of Oral Pathology, SJM Dental College and Hospital, Chitradurga, India
2 Department of Oral Pathology, AB Shetty Memorial Institute of Dental Sciences, Deralakatte, Mangalore, India
3 Department of Oral Pathology, KVG Dental College and Hospital, Sullia, Karnataka, India

Date of Web Publication13-Dec-2013

Correspondence Address:
Kumaraswamy L. R. Naik
Department of Oral Pathology, SJM Dental College and Hospital, Chitradurga, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-9596.122986

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  Abstract 

Dental literature is replete with cases of supernumerary mandibular premolar and unicystic ameloblastoma as separate entities but to our knowledge the present clinical scenario of unicystic ameloblastoma associated with impacted supernumerary premolar was rarely reported. The purpose of this article is to report a patient who presented with pain and swelling of the left mandible of five months' duration. Following clinical evaluation and radiological examination a diagnosis of a rare case of mandibular supernumerary premolar associated with unicystic ameloblastoma was made. The tumor was excised and mandibular reconstruction done.

Keywords: Impacted supernumerary premolar, unicystic ameloblastoma, Supernumerary tooth


How to cite this article:
Naik KL, Shetty P, Shaila M, Sreelatha S V, Chowdappa HT. Unicystic ameloblastoma associated with impacted supernumerary premolar: An unusual case report. Arch Int Surg 2013;3:198-200

How to cite this URL:
Naik KL, Shetty P, Shaila M, Sreelatha S V, Chowdappa HT. Unicystic ameloblastoma associated with impacted supernumerary premolar: An unusual case report. Arch Int Surg [serial online] 2013 [cited 2024 Mar 1];3:198-200. Available from: https://www.archintsurg.org/text.asp?2013/3/2/198/122986


  Introduction Top


Supernumerary teeth (ST) are mammalian developmental abnormalities characterized by the presence of extra teeth in addition to normal eruption series. Prevalence of ST varies between 0.3-1.9% in primary dentition and 1-3.6% in permanent dentition. [1] It has been reported that the prevalence of supernumerary premolar (SP) is rare and is between 0.029 and 0.64%. [2] The term unicystic ameloblastoma (UCA) refers to cystic lesions that show clinical, radiographic, or gross features of a jaw cyst, but on histologic examination show a typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal or mural tumor growth. [3] Most of the UCAs are associated with an impacted tooth, which is usually the mandibular third molar. [4] Here we report a rare case of UCA associated with an impacted mandibular SP.


  Case Report Top


A 19-year-old male visited the department of oral medicine and radiology, A.B. Shetty Memorial Institute of Dental Sciences with a complaint of painful swelling in lower left side of the jaw since 5 months. The swelling gradually increased to reach the present size. Pain was moderate, intermittent, localized and presented only on chewing or biting hard food. Patient also experienced slight numbness in the left lower corner of the lip area since 3 months. On extraoral examination gross facial asymmetry was seen due to the presence of a solitary diffuse swelling in the left lower third of the face, measuring about 4 × 6 cm [Figure 1]. On palpation, local rise in temperature and tenderness was noticed. Swelling was firm to hard in consistency. Left solitary submandibular lymph node was palpable. It was mobile, firm in consistency and measured 3 × 3 cm. On intraoral examination a swelling in the left buccal sulcus obliterating the buccal vestibule was seen, measuring 3 × 1 cm, extending from the mandibular second premolar to the third molar region. Swelling was firm in consistency and tender on palpation. Left mandibular premolars, first and second molar showed Grade I mobility and delayed response to vitality test. On aspiration purulent fluid was seen. Provisional diagnosis of infected odontogenic cyst with differential diagnosis of ameloblastoma was made. From panoramic radiograph, a well-defined unilocular radiolucency extending from the distal aspect of the lower left first premolar to the ascending ramus was demonstrated [Figure 2]. Cortical expansion and thinning of the cortex at the inferior border and the angle of the mandible was seen. A horizontally impacted tooth with incomplete root formation at the level of the first molar root was associated to the lesion. Root resorption of the lower left premolar and molar teeth was shown. Mandibular occlusal view showed buccal cortical plate expansion and horizontally placed ST superimposing the crown of the first molar [Figure 3]. Radiographic differential diagnoses of dentigerous cyst, ameloblastoma, odontogenic keratocyst and calcifying epithelial odontogenic cyst were considered. Incisional biopsy revealed a cystic lesion with reduced enamel epithelial (REE) lining. Connective tissue capsule was made up of dense bundles of collagen fibers with moderate infiltration of chronic inflammatory cell infiltrate. Area of odontogenic epithelium proliferating into the capsular connective tissue in the form of follicular ameloblastoma was noted [Figure 4]. Diagnosis of UCA with intra-mural proliferation was made. Segmental en bloc resection of left mandible was done preserving the inferior border of the mandible. Inferior alveolar nerve was preserved. Gross surgical specimen consisted of resected mandible with horizontally impacted SP with a cystic lesion. The specimen measured 5 × 4 × 3 cm [Figure 5]. Cystic mass was surrounding the crown of an impacted tooth. On sectioning, a brownish black intraluminal soft-tissue mass measuring 2 × 1 × 2 cm was noted. Excisional biopsy features were similar to that of incisional biopsy. In addition, sections from the luminal soft-tissue mass also revealed similar features of follicular ameloblastoma. Final diagnosis of UCA with mural and intraluminal proliferation associated with impacted SP was made. Follow-up of the patient for ten years was uneventful.
Figure 1: Extraoral photograph showing diffuse swelling in the left lower one-third of the face

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Figure 2: Panoramic radiograph showing well-defi ned unilocular radiolucency with impacted supernumerary premolar on left side of mandible

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Figure 3: Mandibular occlusal radiograph showing buccal cortical plate expansion

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Figure 4: Photomicrograph showing ameloblastoma follicles proliferating into the capsular connective tissue (H and E, ×10)

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Figure 5: Photograph of surgical specimen consisted of resected mandible with horizontally impacted supernumerary premolar with a cystic lesion. Also see the soft tissue mass removed from the cystic lumen

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  Discussion Top


UCA is a rare type, accounting for about 6% of ameloblastomas. It occurs usually in the younger age group of 16-20 years. The gender distribution shows a slight male predilection with a male to female ratio of 1.6:1, when associated with an unerupted tooth. [5] Clinical and radiographic findings in most cases of UCA suggest that the lesion is an odontogenic cyst, in particular dentigerous cyst. [4] UCA is usually asymptomatic, although a large tumor may cause painless swelling of the jaws with facial asymmetry. [5] In our case the patient complained of pain due to secondary infection of the lesion. Radiographic appearance of UCA is usually unilocular. Involved teeth in the area show varying degrees of root resorption. [6] The epithelial lining of the UCA is not always uniformly characteristic. It is often lined partly by a nonspecific thin epithelium, usually of REE. [4] All these features were seen in the present case. Whether UCA originates de novo as a neoplasm or it is a result of neoplastic transformation of non-neoplastic cyst epithelium has long been debated. [7] A definitive diagnosis of UCA can only be done by histological examination of the entire lesion and cannot be predicted preoperatively on clinical or radiographic grounds. As preoperative incisional biopsy is not representative of the entire lesion, it may result in an incorrect diagnosis unless a representative site is available as in the present case. When in doubt it is advised to go for further advanced imaging such as computed tomography or magnetic resonance imaging with contrast enhancement. [8],[9] Present case showed both mural and intraluminal proliferation of ameloblastic islands. Hence it can be classified as Group III according to Ackermann and subgroup 1.3 according to Phillipsen and Reichart. [9] Treatment depends on the histological type and UCA with mural proliferation should be treated aggressively. [4] Recurrences were reported in up to 3.6%, hence long-term follow-up is advised. [3]

SPs occur 3 times more in males than in females. [9] Present case was seen in a male patient. Mandibular SPs are late in their development. [10] In our case there was incomplete root formation though the patient was 19 years old. Cases of familial ST have been described though the present case was not associated with such familial history. [10] According to the literature, only 2% of the SPs are likely to undergo pathological changes. Cyst formation and root resorption have been cited in the literature as frequent complications. [2] Recurrence of SP after being excised is not uncommon, however, the present case did not show any such recurrences. [11]


  Conclusion Top


A rare case of simultaneous occurrence of mandibular SP associated with UCA is reported. Impacted supernumerary teeth are usually found on routine radiography unless associated with any pathology. Though the percentage of pathologies associated with impacted SPs is less, in our opinion, if feasible, impacted supernumeraries should be removed to prevent future morbidity. Treatment of UCA depends on the histological type and long-term follow-up is advised.

 
  References Top

1.Saini T, Keene JJ, Whetten J. Radiographic diagnosis of supernumerary premolars: Case reviews. J Dent Child 2002;69:84-90.  Back to cited text no. 1
    
2.Kasat VO, Saluja H, Kalburge JV, Kini Y, Nikam A, Laddha R. Multiple bilateral supernumerary mandibular premolars in a non-syndromic patient with associated orthokeratinized odontogenic cyst: A case report and review of literature. Contemp Clin Dent 2012;3:248-52.  Back to cited text no. 2
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3.Li TJ, Wu YT, Yu SF, Yu GY. Unicystic ameloblastoma: A clinicopathologic study of 33 Chinese patients. Am J Surg Pathol 2000;24:1385-92.  Back to cited text no. 3
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4.Nagalaxmi V, Sangmesh M, Maloth KN, Kodangal S, Chappidi V, Goyal S. Unicystic mural ameloblastoma: An unusual case report. Case Rep Dent 2013;2013:957418.  Back to cited text no. 4
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5.Kumar KR, George GB, Padiyath S, Roopak S. Mural unicystic ameloblastoma crossing the midline: A rare case report. Int J Odontol Stomatol 2012;6:97-103.  Back to cited text no. 5
    
6.Eversole LR, Leider AS, Strub D. Radiographic characteristic of cystogenic ameloblastoma. Oral Surg Oral Med Oral Pathol 1984;57:572-7.  Back to cited text no. 6
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7.Robinson L, Martinez MG. Unicystic ameloblastoma: A prognostically distinct entity. Cancer 1977;40:2278-85.  Back to cited text no. 7
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8.Cankurtaran CZ, Branctetter BF, Chiosea SI, Barnes EL. Ameloblastoma and dentigerous cyst associated with impacted mandibular third molar tooth. Radiographics 2010;30:1415-20.  Back to cited text no. 8
    
9.Roos RE, Raubenheimer EJ, Van Heerden WF. Clinico-pathological study of 30 unicystic ameloblastomas. J Dent Assoc S Afr 1994;49:559-62.  Back to cited text no. 9
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10.O'Dowling I. Supernumerary pre-molar teeth in the mandible. J Ir Dent Assoc 2009;55:293-5.  Back to cited text no. 10
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11.Solares R, Romero MI. Supernumerary premolars: A literature review. Pediatr Dent 2004;26:450-8.  Back to cited text no. 11
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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