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Table of Contents   
CASE REPORT  
Year : 2020  |  Volume : 31  |  Issue : 6  |  Page : 975-978
CBCT evaluation and surgical management of an unusual cystic lesion: A case report


Department of Pediatric and Preventive Dentistry, Amrita School of Dentistry, Amrita Institute of Medical Sciences, Amrita University, Ponnekara, Kochi, Kerala, India

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Date of Submission06-Jun-2019
Date of Decision31-Jan-2020
Date of Acceptance12-Feb-2020
Date of Web Publication22-Mar-2021
 

   Abstract 


This is a case of swelling originating from dens invaginatus associated with lateral incisor, misdiagnosed as a globulomaxillary cyst. The initial diagnosis of globulomaxillary cyst was made solely from radiographs by observing a reverse pear-shaped radiolucency between the lateral incisor and cuspid. The results following pulp testing and Cone Beam Computed Tomography (CBCT) evaluation revealed the endodontic origin of the lesion, which entailed the treatment carried out by surgical enucleation and apicoectomy. This also details the clinical and diagnostic dilemma as well as conflicts associated with the diagnosis and management of the cystic lesion.

Keywords: CBCT, dens invaginatus, diagnostic pitfall, endodontic origin, globulomaxillary cyst

How to cite this article:
Raj R, Veeraraghavan R, Kumaran P, Xavier AM, Varma BR, Menon MM. CBCT evaluation and surgical management of an unusual cystic lesion: A case report. Indian J Dent Res 2020;31:975-8

How to cite this URL:
Raj R, Veeraraghavan R, Kumaran P, Xavier AM, Varma BR, Menon MM. CBCT evaluation and surgical management of an unusual cystic lesion: A case report. Indian J Dent Res [serial online] 2020 [cited 2021 Sep 22];31:975-8. Available from: https://www.ijdr.in/text.asp?2020/31/6/975/311655



   Introduction Top


The classical description of 'globulomaxillary cyst' as a typical fissural cyst, induced by entrapment of epithelial cells amidst the globular and maxillary process,[1] has been questioned for its existence. The retrospective study of Dammer et al. (2013) had disapproved the concept of 'globulomaxillary cyst' as an entity of its own. Instead, it may be used to anatomically hypothesise a lesion that appears between the maxillary lateral incisor and canine, and a final diagnosis should be confirmed only after a thorough comprehensive clinical, radiological and histopathological examination.[2]

Advanced imaging techniques like Cone Beam Computed Tomography (CBCT) can be helpful to unveil the diagnosis of cystic lesions in our day-to-day practice that resemble or correspond to the earlier picture of globulomaxillary cyst.


   Case Report Top


A 14-year-old female patient was referred from an outside clinic with the complaint of pain and swelling on the upper right front region for the past one week. The pain was sudden in onset, and an insidious peanut-sized swelling had appeared on the right side of the face over this past one week.

Extra-oral examination revealed a solitary, ill-defined spherical swelling in the right middle third of the face, measuring approximately 1.5 cm in diameter, extending from the philtrum to the right corner of mouth anteroposteriorly and from ala of the nose to vermillion border of upper lip line superoinferiorly [Figure 1]a. The overlying skin was healthy, with no local rise in temperature. On palpation, the swelling was bony hard in consistency, tender and immobile.
Figure 1: Pre-operative findings and investigations. a) Patient's photograph showing extra-oral swelling; b) Intra-oral swelling with vestibular obliteration; c) Intra-oral Periapical Radiograph (IOPAR); d) Fine-Needle Aspiration of Cyst; e-g) CBCT Images

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Intra-oral examination revealed a solitary diffuse swelling concerning 12 and 13 regions, measuring approximately 1 cm in diameter, extending from mesial aspect of 13 to distal aspect of 13 mesiodistally and superoinferiorly from the depth of labial vestibule to free marginal gingiva of 13 with marked obliteration of the vestibule [Figure 1]b. On inspection, a slightly red, smooth swelling with no pulsations was observed. Palpation revealed the ovoid-shaped swelling to be tender, afebrile and with a soft consistency.

The extensive clinical examination led to a provisional diagnosis of benign cystic lesion of odontogenic or non-odontogenic origin. The upper right lateral incisor tested to be non-vital. An intra-oral periapical radiograph revealed well-defined reverse pear-shaped radiolucency extending up to the periapical region of the tooth with displacement of roots associated with right maxillary lateral incisor and canine [Figure 1]c. The radiograph also showed an intact lamina dura concerning the canine and an open apex concerning the lateral incisor. The classical pear-shaped radiolucency in the lateral incisor–canine region pointed out the possibility of a globulomaxillary cyst. Fine-needle aspiration of the swelling showed a discharge containing pus and blood [Figure 1]d. A detailed investigation with CBCT 5 × 5 section showed a well-defined uniform hypodense area measuring approximately 18.3 × 10.5 mm between the lateral incisor and canine regions [Figure 1]e and [Figure 1]f. Cortical thinning was present buccally and palatally with possible erosion on the palatal side in some slices. A remarkable finding in the sagittal section of CBCT was a pear-shaped invagination of enamel and dentin with a narrow constriction at the opening of the surface of the incisor approximating pulp, suggestive of Dens invaginatus [Figure 1]g. The radiographic diagnosis confirmed a periapical cyst in relation to the lateral incisor.

Following the diagnosis, the treatment commenced with apexification of right maxillary lateral incisor. After standard betadine preparation and isolation, a crevicular incision was placed from canine to canine with the release incision distal to the canine [Figure 2]a. A raised mucoperiosteal flap exposed the bone, and the persisting bony defect [Figure 2]b, made it possible for enucleation of the cystic specimen of 5 × 5 dimension [Figure 2]c. Apicoectomy of 12 and retrograde filling with Glass Ionomer Cement (GIC) completed the treatment. Another observation on elevating the flap was resorption of the bone covering (cortical) the buccal aspect of the root of the canine. It was decided to monitor the vitality of the canine and proceed with root canal treatment, only if symptomatic. Histopathological examination showed a central cystic lumen surrounded by inflammatory infiltrates and macrophages confirming our radiographic diagnosis of the periapical cyst [Figure 2]d. Post-surgical period was uneventful, and the patient was recalled at intervals of 7 days, 14 days, 3 months and 6 months [Figure 3]a, [Figure 3]b and [Figure 3]c. The three-month review showed change in the vitality of the canine, which was followed by root canal treatment [Figure 3]d and [Figure 3]e.
Figure 2: Surgical enucleation, apicoectomy procedure and histopathology. a) Surgical enucleation; b) Apicoectomy; c) Cystic specimen; d) Histopathologic findings

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Figure 3: Periodic recall and follow-up. a) Post-surgical photograph and b) and c) Intra-oral photograph and IOPAR at 3 months recall. d) and e) IOPAR and photograph after root canal treatment of 13

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   Discussion Top


The early portrayal of globulomaxillary cyst by Thoma et al. in 1937 has often been the subject of dubious controversies regarding its embryological origin and its adequacy to be considered as a sole entity.[1] Various authors like Christ (1970), Wysocki and Goldblatt (1981) and Shear (1992)[3] considered them as an alternative entity through their retrospective study by evaluating a few reports of 'globulomaxillary cysts', and the denouement of their reviews revoked the 'globulomaxillary cyst' as a discrete entity.[4],[5] 'Globulomaxillary cysts' keep being depicted in the literature or conferred in a differential diagnostic perspective even in our day-to-day practice regardless of these findings.

D'Silva and Anderssan (1993) postulated that cells of epithelial origin may get trapped between the lateral incisor and canine during the embryological fusion of facial process. This theory led them to propose that the globulomaxillary cyst should be still contemplated as a separate entity from an embryological and histopathological viewpoint.[6] Chimenti et al. (1996) acknowledged the 'fissural' histogenetic hypothesis of its origin by discussing five new cases of globulomaxillary cysts.[7]

Häring et al. (2006) through their extensive review concluded that the term 'globulomaxillary' must be used only to anatomically hypothesise a lesion occurring between the maxillary lateral incisor and the adjacent canine, and a series of comprehensive clinical, radiological and histopathological examinations are needed to arrive at a definitive diagnosis. They also highlighted the fact that the 'globulomaxillary cyst' did not appear in the recently published WHO classification of head and neck tumours (2005) and, therefore, it is to be presumed that this form of cyst is no longer accepted as an entity of its own.[8]

The results of retrospective study by Dammer et al. (2013) along with vast amount of scientific studies on this topic would dispute the continuation of a globulomaxillary cyst as Thoma described it in 1937.[2] However, in daily practice, osteolytic alteration in globulomaxillary region is still reported with a higher than average frequency (0.7%) of all cysts of jaw (Saran et al., 2016; Reddy et al. 2017).[9],[10] Mistreatment in particular, as regards to the teeth confining the cyst, is often a possible sequela of ignorance of the correct diagnosis. The clinician should hypothesise an odontogenic origin for the lesion, when such radiolucencies are encountered in the maxillary lateral incisor and canine region.[11] In the present case, histopathological examination pointed out the absence of any definitive epithelial origin, which ruled out the possibility of globulomaxillary cyst or any developmental cyst. Advanced imaging techniques like Cone Beam Computed Tomography (CBCT) can give adequate three-dimensional views of oral and maxillofacial structures. In this particular case, CBCT assisted in the pre-operative diagnosis of the radicular cyst, thus helping in surgical planning.[12]


   Conclusion Top


Shreds of evidence from past studies suggest that the majority of cysts that occur in this particular region are of odontogenic origin.[5] Nevertheless, non-odontogenic cysts do still occur in this region in our daily practice. An odontogenic cyst, a giant cell granuloma, adenomatoid odontogenic tumour, surgical defects, myxomas, anterior bony clefts and especially anatomic variations may masquerade as a globulomaxillary cyst.[13] In addition, this case report highlights the significance of CBCT in aiding with diagnosis of the condition because of its high spatial resolution and undistorted hard tissue formation. However, further research studies are warranted to confirm the exact aetiology and prevalence of globulomaxillary cysts.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images atnd other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Thoma KH. Facial cleft or fissural cysts. Int J Orthod Oral Surgery 1937;23:83-9.  Back to cited text no. 1
    
2.
Dammer U, Driemel O, Mohren W, Giedl C, Reichert TE. Globulomaxillary cysts-”Do they really exist? Clin Oral Investig 2014;18:239-46.  Back to cited text no. 2
    
3.
Shear M. Cysts of the oral regions. Third edn. Butterworth- Heinemann Ltd, London, UK. 1992.  Back to cited text no. 3
    
4.
Christ TF. The globulomaxillary cyst: An embryologic misconception. Oral Surg Oral Med Oral Pathol 1970;30:515-26.  Back to cited text no. 4
    
5.
Wysocki GP, Goldblatt LI. The so-called “globulomaxillary cyst” is extinct. Oral Surg Oral Med Oral Pathol 1993;76:185-6.  Back to cited text no. 5
    
6.
D'silva NJ, Anderson L. Globulomaxillary cyst revisited. Oral Surg Oral Med Oral Pathol 1993;76:182-4.  Back to cited text no. 6
    
7.
Chimenti C, Monaco A, Nardi E, Volpe F. Globulomaxillary cysts. Minerva Stomatol 1996;45:589-92.  Back to cited text no. 7
    
8.
Häring P, Filippi A, Bornstein MM, Altermatt HJ, Buser D, Lambrecht JT. The “globulomaxillary cyst” a specific entity or a myth? Schweiz Monatsschr Zahnmed 2006;116:380-97.  Back to cited text no. 8
    
9.
Saran G, Umapathy D, Mishra N, Channaiah SK, Rai P. Globulomaxillary cyst in a 16-year-old male patient-A case report with review. J Oral Surg Oral Med Oral Pathol Oral Radiol 2016;2:164-6.  Back to cited text no. 9
    
10.
Reddy LS, Jain V, Mittal S, Gupta SD. Full moon-shaped unusual appearance of a globulomaxillary cyst: A case report. J Indian Acad Oral Med Radiol 2017;29:50-2.  Back to cited text no. 10
  [Full text]  
11.
Kumar LS, Manuel S, Nair BJ, Nair V. An ambiguous asymptomatic swelling in the maxillary anterior region-”A case report. Int J Surg Case Rep 2016;23:65-9.  Back to cited text no. 11
    
12.
Venkatesh E, Elluru SV. Cone-beam computed tomography: Basics and applications in dentistry. J Istanbul Univ Fac Dent 2017;51 (3 Suppl 1):S102-21.  Back to cited text no. 12
    
13.
Wood NK, Goaz PW. Differential Diagnosis of Oral and Maxillofacial Lesions. 5th ed. New Delhi: Elsevier Publishers; 1997. p. 300-1.  Back to cited text no. 13
    

Top
Correspondence Address:
Dr. Parvathy Kumaran
Department of Pediatric and Preventive Dentistry, Amrita School of Dentistry, Amrita Institute of Medical Sciences, Amrita University, Ponekara, Kochi, Kerala - 682 041
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_470_19

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  [Figure 1], [Figure 2], [Figure 3]



 

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    Abstract
   Introduction
   Case Report
   Discussion
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