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Calcifying Epithelial Odontogenic Tumor or Pindborg tumor : about a case in CHU-JDR 

Antananarivo. 

 

Fahasoavana Rohamah Njatosoa¹*, Henri Rantonirina Andriamanantena  ², Fenosoa Vonimbola 

d’AssiseRakotoarimanana ², Emmanuel Andrianony Rakotoarivony ¹, Aurélien Richard 

Rakotoarison ³,  Alberto John Bam Razafindrabe ². 

¹ Department of Maxillo-facial surgery in Hospital Centrer Professor Zafisaona Gabriel Androva, 
Mahajanga, Madagascar 
² Department of maxillo-facial surgery in Hospital Centrer University of Joseph Dieudonné 
Rakotovao (CHU-JDR) – Befelatanana Antananarivo 
³ Department of Oto-Rhino-Laryngology and Cervico-Facial in (CHENOSOA) Hospital Centrer 

of  Soavinandriana Antananarivo 

 

   
  

                                          
  

    
 

Abstract 

 
Pindborg tumor or Calcifying Epithelial Odontogenic Tumor (CEOT) derived from the 

enamel epithelium. It is benign, rare (less than 1% of odontogenic tumors), painless, slowly 

evolving, hardand is embedded in the bone. It is frequent in subjects between 20 and 60 years 

old, with a balanced sex ratio. The area of predilection is the mandibular bone. The objective 

of this study was to diagnose Pindborg tumor and to determine its management. We report the 

case of a 58-year-old woman, evolving for 10 years in the left ramus. The tumor was removed 

by enucleation under general anesthesia, then a recurrence occurred after three years. It was 

painless, bone-like, firm, benign looking, filling the vestibule. CT scan showed a mixed 

density osteolytic and osteocondensing image in the left ramus, blowing out the cortical bone, 

partitioned and seat of multiple calcifications. The second procedure consisted of an 

interrupting mandibular resection and immediate placement of a titanium screw plate for 

reconstruction.This tumor may be confused with ameloblastoma, ossifying fibroma, and other 

IJO JOURNALS

Volume 07 | Issue 01 | January 2024 | https://ijojournals.com/index.php/hsn/index 1

correspondant author : Fahasoavana Rohamah NJATOSOA
                               Department of Maxillo-facial surgery in Hospital Centrer Professor

  Zafisaona Gabriel Androva, Mahajanga, Madagascar 

( ISSN: 2814-2098 )

https://ijojournals.com/

Fahasoavana Rohamah Njatosoa¹*

                                                     Volume 07 || Issue 01 || January., 2024 ||

mailto:njatosoas@outlook.com/


IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  
 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 

http://ijojournals.com/            Volume 07 Issue 01 || January., 2024 || 

 

 

 

 
 

odontogenic tumors with mineralized components. The anatomopathological examination of 

the surgical specimen confirmed the diagnosis of CEOT. Because of its compartmentalized 

form, interrupted bone resection is the best technique to avoid recurrence.  

Keywords: benign tumor, CEOT, interrupting resection, recurrence. 

Introduction 

Pindborg's Tumor or Calcifying Epithelial Odontogenic Tumor (CEOT) was first discovered 

by Jens Jorgen Pindborg in 1955 (1). It is a benign tumor, rare (less than 1% of odontogenic 

tumors), painless and slow-growing. It is common in people aged between 20 and 60, with a 

balanced sex ratio (2). Hard, poorly limited, often associated with a molar and integral with 

the bone, the preferred site is mandibular (3). The aim of this study is to report a case of 

TOEC in a 58-year-old woman from Antananarivo. 

Case report 

A 58-year-old woman was referred to us in September 2019 with a recurrent mandibular, 

symphyseal and left paraspinal swelling (figure 1.A). The first tumoral manifestation had 

been evolving for around ten years, involving only the left paraspympetal region, anterior to 

35. Enucleation was performed under general anaesthetic. Three years after the operation, a 

swelling reappeared at the same site, extending into the symphyseal region. The swelling was 

painless, close to the mandibular bone, firm, filling the vestibule, covered with ulcero-necrotic 

mucosa and a whitish coating, and did not bleed on contact. The floor of the mouth was free 

but pushed back, with a healthy mucosa; the tongue was mobile (figure 1.B). The patient had 

no associated sensory-motor disorders, and the cervical lymph nodes were free. The 

panoramic radiograph or orthopantomogram showed a polygeodic image of the left ramus, 

with a large symphyseal-parasymphyseal osteolytic zone and osteocondensing zones 

posteriorly (figure 2). 

Computed tomography confirmed this radiographic diagnosis and added the notion of cortical 

effraction and the presence of multiple calcifications (figure 3). This reappearance of a new 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  
 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 

http://ijojournals.com/            Volume 07 Issue 01 || January., 2024 || 

 

 

 

 
 

lesion at the same site is a probable sign of recurrence.A second operation was performed, 

consisting of an interrupted mandibular resection removing the tumour, followed by 

immediate placement of a titanium screw-plate for reconstruction (figure 4).The post-

operative course was complicated by a submental salivary fistula which disappeared after 

removal of the sutures (figure 5).Pathological examination of the surgical specimen suggested 

a calcified epithelial odontogenic tumor (Pindborg tumor). The excision margins were 

healthy. 

Comments 

CEOTis a tumor usually seen in adults, but it can also develop in children (4). 

Because of their almost identical nature, it is difficult to distinguish CEOT from other benign 

odontogenic tumors such as ameloblastoma, calcifying odontogenic cyst and ossifying 

fibroma (5). Indeed, CEOT and ameloblastoma are two intraosseous tumors that share the 

same clinical and radiological appearance.Their location in the mandibular premolar region is 

similar, as is their slow progression. On CT scan, the images are almost identical: CEOT 

shows a mixed osteolytic and osteocondensing density in the mandibular body, 

compartmentalized and the site of multiple calcifications (6), as found in our patient. In 

ameloblastoma, multilocular soap-bubble osteolysis and multiple root resorption are typical 

(7). The histological appearance of CEOT is specific: cells with enlarged, squamous-like, 

multi-nucleated nuclei and a low mitotic index, as well as amyloid deposits and calcifications 

(8); this is confirmed in our case. Ameloblastoma, on the other hand, derives from Malassez 

epithelial remnants and shows no calcifications (9). 

The indication for treatment depends on the size of the tumour, its relationship with the bony 

cortices, and whether or not it leads to disruption of the basilar border of the mandible, the 

posterior border or the upper end of the ramus (10). Conservative enucleation-type treatment 

is indicated for less aggressive early lesions. This is the case for the management of a 

calcifying odontogenic cyst or a small ossifying fibroma. For invasive, polygeodic, recurrent 

and voluminous tumours such as our case, surgical treatment is radical (9): interrupting bone 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  
 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 

http://ijojournals.com/            Volume 07 Issue 01 || January., 2024 || 

 

 

 

 
 

resection to avoid recurrence.It is important to respect an exeresis margin of 1 to 2cm, in 

healthy tissue, while preserving the functions of neighbouring tissues.  

Thus, the indications for radical or conservative treatment of CEOT depend on the size and 

location of the lesion, and whether it is polygeodic or not.  Indeed, the risk of recurrence after 

enucleation is 15 to 30% at a follow-up of 2 to 4 years (8).Interruptive bone resection is 

indicated for large, recurrent and invasive CEOT(9).However, this technique is highly 

invasive, leading to functional and aesthetic sequelae.  Immediately after tumor removal, a 

titanium screw-plate is inserted to provide a temporary solution. Adaptation of the 

manducatory apparatus required re-education, so that the patient was gradually able to 

mobilize her jaw.The salivary fistula disappeared spontaneously after removal of the 

sutures.Prognosis was good. 

Conclusion 

CEOT is a rare benign tumor specific to dento-maxillary structures.It could be confused with 

ameloblastoma or other odontogenic tumors, and it is the histological examination of the 

lesion that confirms the diagnosis.CEOT shares the same surgical treatment technique as 

ameloblastoma. Bone resection with a good margin off the tumor is the best treatment. 

References 

1. Ide F, Matsumoto N, Kikuchi K, Kusama K. Who Originally Described Pindborg Tumor? 
Head and Neck Pathol. sept 2019;13(3):485-6. 

 
2. Caliaperoumal S, Gowri S, Dinakar J. Pindborg tumor. Contemp Clin Dent. 2016;7(1):95. 

 

3. Priya S, Madanagopaal LR, Sarada V. Pigmented Pindborg tumor of the maxilla: A case 
report. J Oral Maxillofac Pathol. 2016;20(3):548. 

 
4. 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 
and Neck Pathol. déc 2019;13(4):580-6. 
 

5. More CB, VijayvargiyaR. Intraosseous calcifying epithelial odontogenic (Pindborg) 
tumor: A rare entity. J Oral Maxillofac Pathol. 2015;19(2):269. 

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https://pubmed.ncbi.nlm.nih.gov/?term=Vijayvargiya+R&cauthor_id=26604515


IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  
 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 

http://ijojournals.com/            Volume 07 Issue 01 || January., 2024 || 

 

 

 

 
 

 
6. Hafian H, Mauprivez C, Furon V, Pluot M, Lefevre B. Tumeur de Pindborg : à propos 

d’une forme peu différenciée et sans calcification. Revue de Stomatologie et de Chirurgie 
Maxillo-faciale. sept 2004;105(4):227-30. 
 

7. Oikarinen VJ, Boris Calonius PE, Meretoja J. Calcifying epithelial odontogenic tumor 
(Pindborg tumor) Case report. International Journal of Oral Surgery. août 
1976;5(4):187-91. 
 

8. Alraei K, Sharqawi J, Harcher S, Ghita I. Efficacy of the Combination of rhBMP-2 with 
Bone Marrow Aspirate Concentrate in Mandibular Defect Reconstruction after a Pindborg 
Tumor Resection. Case Reports in Dentistry. 18 mars 2020;2020:1-6. 

 
9. Chrcanovic BR, Gomez RS. Calcifying epithelial odontogenic tumor: An updated 

analysis of 339 cases reported in the literature. Journal of Cranio-Maxillofacial Surgery. 
août 2017;45(8):1117-23. 
 

10. Hamdi H, Ben Alaya T, Ben Romdhane I, Khatteche B, Favre de Thierrens C. La tumeur 
épithéliale odontogénique calcifiée : à propos d’un cas. Med Buccale Chir Buccale. 
2002;8(2):87-90. 

 
 

 

Figure 1: A. Facial asymmetry caused by left mandibular tumor. 
     B. Endobuccal aspect of the tumor. 
                (Andriamanantena RH, 2017) 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  
 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 

http://ijojournals.com/            Volume 07 Issue 01 || January., 2024 || 

 

 

 

 
 

 

Figure 2: Orthopantomogram showing an image of a large radiolucent zone in the left symphysis-parasymphysis, and 
polygeodic osteocondensing zones posteriorly with basilar rim involvement.  
(Andriamanantena RH, 2017) 

 

 

Figure 3: Computed tomography (CT) scan showing mixed osteolytic and osteocondensing density, 
compartmentalized and the site of multiple calcifications.  

            A: CT scan with contrast medium injection. 
            B: CT scan without contrast medium injection. 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  
 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 

http://ijojournals.com/            Volume 07 Issue 01 || January., 2024 || 

 

 

 

 
 

(Andriamanantena RH, 2017) 

 

 

Figure 4: Interrupting mandibular resection removing the tumor, followed by placement of a screwed titanium 
reconstruction plate. 
(Andriamanantena RH, 2017) 

 

 

Figure 5: Postoperative follow-up (one month post-op)  
(Andriamanantena RH, 2017) 

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