Home | About us | Editorial board | Ahead of print | Current issue | Archives | Search | Submit article | Instructions | Subscribe | Advertise | Contact us |  Login 
National Journal of Maxillofacial Surgery
 
Print this page Email this page Small font sizeDefault font sizeIncrease font size
Users Online: 544
 


 
Table of Contents
CASE REPORT
Year : 2022  |  Volume : 13  |  Issue : 4  |  Page : 108-112  

Epiphora and nasal blockage – An unusual presentation of Pindborg tumor: A case report and literature review


Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, Dharmsinh Desai University, Nadiad, Gujarat, India

Date of Submission31-May-2020
Date of Acceptance04-Sep-2020
Date of Web Publication20-Aug-2022

Correspondence Address:
Dr. Urvi Shah
8, Kesar Residency, Near Prithvi Hotel, LG Corner, Maninagar, Ahmedabad Gujarat
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/njms.NJMS_104_20

Rights and Permissions
   Abstract 


Odontogenic tumors are silent ailments which can affect any individual. One such lesion is a calcifying epithelial odontogenic tumor (CEOT). It is a rare lesion with a locally aggressive nature. A 33-year-old male presented with nasal blockage and continuous flow of tears from the left eye for the past 7–8 months. Clinical examination revealed slightly protruded left eyeball and altered level of eyeballs with continuous watery discharge. Hess chart confirmed normal eye movements. Intraoral findings were not significant. The final diagnosis of CEOT was established based on the histopathological aspects. CEOT is mostly found in the third to fifth decade of life without gender predilection. Presenting symptoms and signs comprise painless expansile mass, although there are reports associated with pain, nasal obstruction, epistaxis, and proptosis. In this article, we would like to present a case of CEOT with epiphora and nasal blockage as the main and only presentation, an uncommon finding. Furthermore, discussing and posing a question of an adequate period of follow-up required to negate the presence of recurrence. One must stay vigilant enough not only to attest a singular symptom to the commonest ailment related to it but also to explore the possibility of the less known. Furthermore, we need to further research in depth to establish a certain duration after which the likeliness of recurrence is to the minimum.

Keywords: Calcifying epithelial odontogenic tumor, epiphora, maxilla, nasal blockage, pathology, Pindborg tumor


How to cite this article:
Shah U, Patel H, Pandya H, Dewan H. Epiphora and nasal blockage – An unusual presentation of Pindborg tumor: A case report and literature review. Natl J Maxillofac Surg 2022;13, Suppl S1:108-12

How to cite this URL:
Shah U, Patel H, Pandya H, Dewan H. Epiphora and nasal blockage – An unusual presentation of Pindborg tumor: A case report and literature review. Natl J Maxillofac Surg [serial online] 2022 [cited 2022 Sep 28];13, Suppl S1:108-12. Available from: https://www.njms.in/text.asp?2022/13/4/108/353923




   Introduction Top


A calcifying epithelial odontogenic tumor (CEOT) is an uncommon entity comprising 1% of all odontogenic tumors. Its behavior is synchronous to that of ameloblastoma, like being locally expansile, can reoccur, and does not metastasize. This tumor is usually seen in patients in their third to fifth decade, with no gender predilection. Lesions are most commonly found in the molar region of the mandible, followed by premolar region, except for Langerhans cell variant. Large CEOTs may require either marginal or segmental resection, but as it is less aggressive than ameloblastoma, small lesions can be treated more conservatively.[1] An overall recurrence rate of roughly 10% has been reported.[2] Because of its slow growth, a follow-up period of 5 years has been suggested.[3]


   Case Report Top


A 33-year-old male patient presented to the oral and maxillofacial surgery department complaining of nasal blockage and continuous flow of tears from the left eye for the past 7–8 months. Clinical examination revealed slightly protruded left eyeball and altered level of eyeballs with continuous watery discharge [Figure 1]. Complete ophthalmologic evaluation of the patient was done. A Hess chart confirmed normal ocular movement with no neurologic defect. A common cause of epiphora is tear duct obstruction which is, in turn, caused by allergies, infection or inflammation, trauma, excessive eye drop usage, and history of radiation. All these reasons were ruled out by subtractive history taking. It arose the suspicion of obstruction due to a growth within maxilla. The patient also complained regarding nasal blockage which raised suspicion of underlying tumor. Further history revealed that the patient had undergone a previous surgery for growth in the maxillary sinus. At present, there was no complaint of paraesthesia. Intraoral findings were insignificant. The lymph node examination was unremarkable. The patient did not have any comorbidities or destructive habits.
Figure 1: Altered eye levels

Click here to view


Six and a half years ago, the patient had presented with a swelling concerning the left posterior maxillary arch. Examination heeded the presence of a firm nontender growth in the left maxilla involving posterior alveolar mucosa and vestibule. The patient had a history of extraction of 26.27 earlier that year. Radiographic examination had revealed the possibility of well-defined radiolucency in relation to 26, 27, and 28 with thinning of the medial and superior walls of the maxillary sinus, showing calcification and tooth within it. The excised lesion with tooth 28 was diagnosed as CEOT.

The histopathological examination of the previous tumor showed discrete islands, strands, and sheets of polyhedral epithelial cells in fibrous stroma with distinct intercellular bridges. Large areas of amorphous, eosinophilic, hyalinized extracellular material with areas of calcifications were observed. Liesegang rings were found too.

Computed tomography scan of the present illness [Figure 2] stated the presence of ill-defined soft-tissue lesion with diffuse areas of dense calcifications with an expansion of sinus and erosion of its anterior, medial, posterior, and superior wall, extending medially into the left nasal cavity causing pressure on the septum. Attenuation of the left lamina papyracea was also evident. The lesion was extending into the infratemporal fossa and superiorly into the orbit and inferiorly caused erosion of maxillary alveolar ridge. Incisional biopsy affirmed our suspicion of recurrence.
Figure 2: Computed tomography scan with three-dimensional reconstruction and axial and coronal views

Click here to view


Conservative surgical resection was planned under general anesthesia, and a modified Weber–Fergusson's approach was used. Intraoperatively, a well-demarcated lesion from the adjacent tissues was noted [Figure 3].
Figure 3: Surgical procedure

Click here to view


On histopathological examination, tumor mass showed that sheets of epithelial cells with mild nuclear pleomorphism and enlargement associated with hyperchromasia were observed. Plenty of hyaline areas admixed with foci of calcification were noted. The final diagnosis made was CEOT [Figure 4].
Figure 4: H and E-stained specimen slide

Click here to view


Postoperative recovery was inconsequential. Epiphora was controlled overtly during recovery phase. No adverse eye signs were noted. The patient is asymptomatic to date [Figure 5]. One-year postsurgery endoscopic sinus examination was done to rule out recurrence and came out to be negative.
Figure 5: Postoperative image after 1 year

Click here to view



   Discussion Top


The CEOT is additionally referred to as Pindborg tumor and is considered a rare pathological entity representing <2.5% of all odontogenic tumors. CEOT is a benign neoplasm with painless, slow-growing expansion of the jaws. The tumor is usually treated surgically. Devising an effective treatment plan, especially in destructive cases of the upper jaw, becomes necessary as recurrence is not rare with such lesions. The lesions in the posterior maxilla represent <25% of all CEOTs.[4] CEOT is associated with a tooth, erupted or unerupted, in 48% of cases. Radiographically, a mixed radiolucent and radiopaque lesion, unilocular or multilocular, is most characteristic.[1] Radiographic features are similar to an ameloblastoma, dentigerous cyst, or other odontogenic tumors. Although typically benign, CEOT tends to invade local structures and has a potential for recurrence.[5]

Franklin and Pindborg reviewed 113 cases. They mentioned that Pindborg tumor usually presents between 30 and 50 years of age with no gender predilection. Ninety-five percent are intraosseous lesions. Prevalence in the molar region to the premolar region is 3:1. Prevalence according to jaws is mandible to maxilla 2:1. The extraosseous cases (5%) described appearing to have a predilection for the anterior region. Although Pindborg tumors are well described in the mandible, descriptions of lesions involving the maxilla are rare.[6],[7]

The typical presenting symptom is painless asymptomatic expansile mass, although there are reports associated with pain, nasal obstruction, epistaxis, and proptosis. The additional and unusual symptom associated with this case was epiphora, which we did not find mentioned anywhere in the literature. Moreover, it was the main complaint of the patient with nasal blockage as a second and none of the other aforementioned entities.

Epiphora implies overflowing of tears due to impairment of lacrimal drainage. It indicates an unbalance between tear production and tear loss. Causes can be anatomic or functional. Nasolacrimal duct obstruction is caused by allergies, infection or inflammation, trauma, excessive eye drop usage, tumor, or history of radiation. In our case, mechanical obstruction of lacrimal apparatus due to pressure exerted by tumor mass led to difficulty in normal drainage of tears produced which is normally drained in the nasal cavity in the inferior meatus through nasolacrimal duct connected to the lacrimal sac situated at the lacrimal fossa of the lacrimal bone. Furthermore, nasal stuffiness was a result of pressure over the septum by the tumor.

The patient had a previous history of such lesion, so he reported to the maxillofacial surgery department, but in instances where such lesion manifests only as epiphora with no other clinically significant symptom, one is likely to entertain the thought of visiting experts of other specialties as, for example, an ophthalmologist. Hence, getting to the core of a greater disease through means of investigating an unusual symptom becomes crucial.

Literature review

An electronic search was conducted without time restriction in March 2020 from the following databases: PubMed/Medline, Science Direct, Cochrane, and Google Scholar. The terms used for the search were: calcifying epithelial odontogenic tumor or Pindborg tumor.[4],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17] Inclusion criteria comprise tumor involving naso-orbito-maxillary complex, patients with adequate follow-up periods, and cases where presenting signs and symptoms are mentioned. Exclusion criteria included CEOT at other sites than posterior maxilla, cases where presenting signs and symptoms are not mentioned or described vaguely, and cases with irregular/no follow-ups. The titles, abstracts, and full reports (when required) of all reports identified through the electronic searches were read independently by the authors.

Twelve articles were obtained which fit our criteria [Table 1]. Data of 14 patients were noted. The male: female ratio found was 1:1.8. Of 14 cases, only 2 cases (14.28%) had presenting symptoms of nasal stuffiness and proptosis of the eye, whereas 9 cases (64.3%) had a chief complaint of swelling. One case (7.14%) was an accidental finding on routine radiograph. Pain was the first symptom in only one case (7.14%). Epiphora was observed in only one case (7.14%), that is, our case. In 35.7%, maxillectomy was performed. The follow-up period ranged from 18 months to 17 years. In 28.6% of cases, recurrence was observed.
Table 1: Presenting symptoms of the patient with maxillary calcifying epithelial odontogenic tumor

Click here to view


In 2020, De Arruda et al. published the case of a 45-year-old female with CEOT of the left maxilla. First diagnosed in 2004, the patient was operated and recurrence was noted in 2009. The patient refused extensive surgery, and later, in 2014, malignant transformation of the lesion was noted and she was treated accordingly.[17] Hence, malignant transformation of such lesions is also possible, but it is rare. However, recurrence and aggressiveness of lesion is also dependent on histologic type.[18],[19],[20]

Hence, we infer that following the set protocols for treatment and maintaining regular follow-up can result in betterment of the patient. Having said that, further studies to decide adequate duration of follow-up are encouraged.

Ethical approval

Ethical approval was not required and patient identifying knowledge was not presented in this report. Ethical approval is waived.

Consent

The written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review on request.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Author's contribution

All authors contributed significantly and in agreement with the content of the article. All authors have read and approved the final version of the manuscript.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Chrcanovic BR, Gomez RS. Calcifying epithelial odontogenic tumor: An updated analysis of 339 cases reported in the literature. J Craniomaxillofac Surg 2017;45:1117-23. doi: 10.1016/j.jcms.2017.05.007. Epub 2017 May 12.  Back to cited text no. 1
    
2.
de Arruda JA, Abreu LG, Silva LV, Schuch LF, Monteiro JL, Arantes DA, et al. Calcifying epithelial odontogenic tumours: Collaborative study of 32 cases and review of literature. Oral Dis 2019;25:192-205. doi: 10.1111/odi.12958. Epub 2018 Sep 9.  Back to cited text no. 2
    
3.
Shanmuga PS, Ravikumar A, Krishnarathnam K, Rajendiran S. Intraosseous calcifying epithelial odontogenic tumor in a case with multiple myeloma. J Oral Maxillofac Pathol 2009;13:10-3.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Foroughi R, Amini Shakib P, Babaei Darzi A, Seyedmajidi M, Jamaatlou N. Calcifying epithelial odontogenic tumor: Report of a recurrent destructive case with review of literature. J Dent (Tehran) 2015;12:78-84.  Back to cited text no. 4
    
5.
Fazeli SR, Giglou KR, Soliman ML, Ezzat WH, Salama A, Zhao Q. Calcifying epithelial odontogenic (Pindborg) tumor in a child: A case report and literature review. Head Neck Pathol 2019;13:580-6.  Back to cited text no. 5
    
6.
Franklin CD, Pindborg JJ. The calcifying epithelial odontogenic tumor. A review and analysis of 113 cases. Oral Surg Oral Med Oral Pathol 1976;42:753-65.  Back to cited text no. 6
    
7.
Gon F. The calcifying epithelial odontogenic tumor. Report of a case and a study of its histogenesis. Brit J Cancer 1965;19:39-50.  Back to cited text no. 7
    
8.
Stimson PG, Luna MA, Butler JJ. Seventeen-year history of a calcifying epithelial odontogenic (Pindborg) tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1968;25:204-8.  Back to cited text no. 8
    
9.
Ai-Ru L, Zhen L, Jian S. Calcifying epithelial odontogenic tumors: A clinicopathologic study of nine cases. J Oral Pathol 1982;11:399-406.  Back to cited text no. 9
    
10.
Lee CY, Mohammadi H, Mostofi R, Habibi A. Calcifying epithelial odontogenic tumor of the maxillary sinus. J Oral Maxillofac Surg 1992;50:1326-8.  Back to cited text no. 10
    
11.
Patiño B, Fernández-Alba J, Garcia-Rozado A, Martin R, López-Cedrún JL, Sanromán B. Calcifying epithelial odontogenic (pindborg) tumor: A series of 4 distinctive cases and a review of the literature. J Oral Maxillofac Surg 2005;63:1361-8.  Back to cited text no. 11
    
12.
Gopalakrishnan R, Simonton S, Rohrer MD, Koutlas IG. Cystic variant of calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:773-7.  Back to cited text no. 12
    
13.
Bridle C, Visram K, Piper K, Ali N. Maxillary calcifying epithelial odontogenic (Pindborg) tumor presenting with abnormal eye signs: Case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:e12-5.  Back to cited text no. 13
    
14.
Sedghizadeh PP, Wong D, Shuler CF, Linz V, Kalmar JR, Allen CM. Multifocal calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:e30-4.  Back to cited text no. 14
    
15.
Channappa NK, Krishnapillai R, Rao JB. Cystic variant of calcifying epitelial odontogenic tumor. J Investig Clin Dent 2012;3:152-6.  Back to cited text no. 15
    
16.
Gruber K, de Freitas Filho SA, Dogenski LC, da Silva Bocassanta AC, Paranhos LR, de Carli JP. Surgical management of a large calcifying epithelial odontogenic tumor in the maxilla: A case report. Int J Surg Case Rep 2019;57:197-200.  Back to cited text no. 16
    
17.
de Arruda JA, Arantes DA, Schuch LF, Mosconi C, Abreu LG, de Andrade BA, et al. A rare case of an aggressive clear cell variant of calcifying epithelial odontogenic tumor in the posterior maxilla. Int J Surg Pathol 2020;28:526-35.  Back to cited text no. 17
    
18.
Veness MJ, Morgan G, Collins AP, Walker DM. Calcifying epithelial odontogenic (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck 2001;23:692-6.  Back to cited text no. 18
    
19.
Kawano K, Ono K, Yada N, Takahashi Y, Kashima K, Yokoyama S, et al. Malignant calcifying epithelial odontogenic tumor of the mandible: Report of a case with pulmonary metastasis showing remarkable response to platinum derivatives. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:76-81.  Back to cited text no. 19
    
20.
Kumar M, Fasanmade A, Barrett AW, Mack G, Newman L, Hyde NC. Metastasising clear cell odontogenic carcinoma: A case report and review of the literature. Oral Oncol 2003;39:190-4.  Back to cited text no. 20
    


    Figures

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

  [Table 1]



 

Top
   
 
  Search
 
    Similar in PUBMED
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures
    Article Tables

 Article Access Statistics
    Viewed110    
    Printed6    
    Emailed0    
    PDF Downloaded22    
    Comments [Add]    

Recommend this journal