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Mandibular reconstruction after Pindborg tumor: case report
HENRIQUE CLASEN SCARPARO 1 | RONIELE LIMA DOS-SANTOS 1 | ALEXANDRE MARANHÃO MENEZES NETO 1 | FRANCISCO SAMUEL RODRIGUES CARVALHO 1 | FABRICIO DE LAMARE RAMOS 1 |
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FÁBIO WILDSON GURGEL COSTA 1 | EDUARDO COSTA STUDART SOARES 1
ABSTRACT
Pindborg tumor, or calcifying epithelial odontogenic tumor (CEOT), is a rare, benign neoplasm with locally aggressive biological behavior. It has higher incidence between the third and fifth decades of life, with no predilection for race or sex. It presents radiographically with mixed pattern containing radiolucent areas with calcified structures. The objective of this study was to present a clinical case of CEOT in a 42-year-old female patient, with a slight increase in volume in the posterior region of the mandible on the left side. An electronic data search was also carried out taking into account the last 10 years using the following electronic databases: PubMed, Cochrane database of systematic reviews, Embase, Medline and Electronic Journal Center. Imaging examinations demonstrated the presence of extensive radiolucent lesion with areas of scattered calcifications. The patient was submitted to an incisional biopsy of the lesion whose diagnosis was of CEOT. The patient was submitted to resection of the tumor, posterior reconstruction of the affected site with autogenous graft and implant-supported prosthesis. The patient was asymptomatic, under clinical-imaging follow-up for 2 years, with no signs of recurrence of the lesion, besides rehabilitation in which satisfactory aesthetic-functional results were obtained.
Keywords: Mandible. Mandibular reconstruction. Mandibular neoplasms.
1 Universidade Federal do Ceará, Hospital Universitário Walter Cantídio, Serviço de Cirurgia e Traumatologia Bucomaxilofacial (Fortaleza/CE, Brazil).
Contact address: Alexandre Maranhão Menezes Neto Rua Capitão Francisco Pedro, 1016, apto. 303 – CEP: 60.430-372 – Fortaleza/CE E-mail: alexandremaranhaobucomaxilo@gmail.com
How to cite: Scarparo HC, Dos-Santos RL, Menezes Neto AM, Carvalho FSR, Ramos FL, Costa FWG, Soares ECS. Mandibular reconstruction after Pindborg tumor: case report. J Braz Coll Oral Maxillofac Surg. 2019 Jan-Apr;5(1):46-52. DOI: https://doi.org/10.14436/2358-2782.5.1.046-052.oar
Submitted: November 06, 2017 - Revised and accepted: April 18, 2018
» The authors report no commercial, proprietary or financial interest in the products or companies described in this article.
» Patients displayed in this article previously approved the use of their facial and intraoral photo graphs.
INTRODUCTION
The calcifying epithelial odontogenic tumor (CEOT), or Pindborg tumor, is a rare benign odontogenic neoplasm. It was first described in 1955 by Jens J. Pindborg. 1 This lesion represents less than 1% of all odontogenic tumors, preferably affecting patients between the third and fifth decades of life, without gender predilection, and occasional cases of malignancy were described in the literature. 2,4 Several studies investigated the pathogenesis of CEOT; however, it remains controversial. 4,5
Its main clinical manifestation is the increase in volume, usually without associated symptomatology. The most affected site is the posterior mandibular region, and approximately 25% of cases occur in the maxilla. 3 It can present in intraosseous (94%) or extraosseous (6%) manifestations, usually with slow and painless growth. 4,6,7
The radiographic presentation is varied and may contain radiolucent areas, with calcified structures of varying sizes and densities. When associated with impacted teeth, these radiopacities are close to the coronal portion. 8,9
Histopathologically, the most common pattern presents polyhedral epithelial cells with well-defined borders and prominent intercellular bridges. Amyloid substances and Liesegang calcified concentric rings are the most classical findings. 10
The Pindborg tumor is locally aggressive. Studies show that, depending on the lesion size, it can be treated by enucleation or by surgical resection with free margins. 10 This procedure may involve conservative enucleation, marginal, segmental or partial resection (larger and more aggressive tumors), or very rarely composite resection (in the presence of malignant transformation). 1,6,7 A local recurrence rate of 14% is reported and the prognosis is considered as good. 3
Therefore, this paper presents a case report of CEOT treated by surgical excision with application of Carnoy solution, posterior reconstruction with autogenous graft and rehabilitation with implants, and also presents a literature review of the last 10 years about the reconstructions used for treatment of this lesion. For that purpose, an electronic data search was conducted on June 2016, considering the last 10 years and using the following electronic databases: PubMed, Cochrane Database of Systematic Reviews, Cochrane Controlled Randomized Controlled Trials, Embase, Medline and Electronic Journal Center. The following search entries were used: “Pindborg”, “calcifying epithelial odontogenic tumor”, “excisional biopsy” and/or “Pindborg”, “treatment” and/or “Pindborg”, “resection” and/or “Pindborg”, “Carnoy solution” and/or “Pindborg”, “recurrence” and/or “Pindborg”. The search included any type of study in which surgical treatment was performed to remove the Pindborg tumor associated with bone reconstruction. Studies published in languages other than English, papers that did not inform the type of treatment and animal studies were excluded. The electronic search retrieved 111 references, among which 37 were animal studies and 8 were not published in English language. Thus, 66 articles were selected for reading. Analysis of abstracts and reading of the full texts retrieved 10 papers that addressed the surgical removal of Pindborg tumor.
CASE REPORT
A 42-year-old female patient, without systemic disorders, attended the dental office for routine examination with the following chief complaint: “I discovered a lump in my mandible three months ago”.
Extraoral physical examination did not show any noticeable changes. Oral examination revealed a painless swelling at the region of teeth 45 to 48, without change in color of the adjacent mucosa (Fig 1). Imaging exam-
inations revealed a multilocular, radiolucent area with well-defined margins and limits, associated with teeth 44, 45 and 47, causing a slight increase in the buccal volume and on the mandibular base (Fig 2A, B). Considering the clinical and radiographic findings, the following diagnosis hypotheses were suggested: CEOT, calcifying odontogenic cyst, ameloblastoma, ossifying fibroma, keratocystic odontogenic tumor and dentigerous cyst.
An incisional biopsy was performed under local anesthesia, and the specimen was referred to an Oral Pathology laboratory. The histopathological analysis revealed polyhedral odontogenic epithelial cells and prominent intercellular junctions with cell pleomorphism, organized as anastomosed strands within a fibrous stroma with hemorrhage areas (Fig 3A). Congo red staining was positive interspersed between the odontogenic epithelial cells, evidencing deposition of eosinophilic material, either globular or amorphous, compatible with amyloid material (Fig 3B), characteristic of the Pindborg tumor.
A biomodel was used for surgical planning and reconstruction plate modeling (Fig. 4A). The patient was then submitted to surgery in a hospital environment, under general anesthesia and by intraoral access. We decided to extract the teeth 44, 45 and 47 because they were affected by the lesion. Surgical resection was performed, including a marginal portion of apparently healthy bone, and Carnoy solution was applied to the surgical site. A 2.0 system plate with high profile was fixed on the base to reinforce the bone remnant (Fig 4B). After 14 days, a second surgery was performed for mandibular reconstruction using an iliac crest bone graft (Fig 5A, B). The patient has been submitted to clinical and imaging follow-up for two years, without signs of lesion recurrence, with rehabilitation by implant-supported denture, which retrieved satisfactory esthetic and functional results (Fig 6A, B).
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Figure 2: A) Cone-beam computed tomography and panoramic reconstruction. B) Three-dimensional reconstruction of computed tomography.
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Figure 3: A) Histopathological section of Pindborg tumor (HE/20x). B) Histopathological section of Pindborg tumor (HE/40X).
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Figure 4: A) 3D surgical biomodel. B) High-profile 2.0 system plate.
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Figure 5: A) Iliac graft positioned with 2.0 system screws. B) Postoperative panoramic radiograph.
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Figure 6: A) Final occlusion. B) Two-year postoperative panoramic radiograph.
DISCUSSION
The Pindborg tumor is an extremely rare odontogenic epithelial neoplasm first described in 1955 by Jens J. Pindborg, 1,7,9,10 representing less than 1% of all odontogenic tumors. Its prevalence affects a broad age range; however, most reported cases comprise patients between the third and fifth decades of life, and it does not present gender predilection. 6,8,9
It manifests with slow and asymptomatic, yet locally invasive growth. Aggressive lesions exhibit bone destruction and tumor calcification. Usually, it is diagnosed during routine dental examination, possibly affecting the maxilla or mandible, yet the most affected site is the posterior mandibular region, predominantly at the premolar and molar regions. 4,6,8
Its most common histopathological characteristic is the presence of pleomorphic polyhedral epithelial cells and prominent intercellular bridges, as well as amyloid substances and Liesegang calcified concentric rings. 1,3,5,6
The differential diagnosis of CEOT include the calcifying odontogenic cyst, ameloblastoma, ossifying fibroma, ameloblastic fibro-odontoma, myxoma, odontogenic tumor keratocystic and dentigerous cyst. 1,8
The type of treatment depends on the size and anatomical location of the CEOT. 6.2 The current literature reports only three cases of malignant CEOT and one case presenting microscopic characteristics of malignant potential, published in the English-lan-
guage literature. 1,4 Odontogenic malignant tumors are extremely rare. Malignancy is confirmed based on the following criteria: histological findings of infiltrative growth, atypical cytologic features and focal necrosis or clear evidence of distant metastatic dissemination. 10
In the present case, the treatment consisted of surgical tumor resection, including the marginal portion of apparently healthy bone, together with reconstruction with plate and iliac crest graft. However, although there are no reports in the literature, Carnoy solution was used in the present patient to assure a greater safety that there would be no lesion relapse. The present search found an article that reported the case of a patient with Pindborg tumor in the posterior mandibular region, in which the same treatment modality was used. However, in the present case, we decided to use an autogenous iliac crest graft, besides subsequent rehabilitation with implants.
Among the reconstruction modalities, autogenous bone is considered among the best options. 5,6 The autogenous graft is biologically considered the gold standard for reconstruction of maxillary and mandibular ridges, because of advantages as antigenic and angiogenic properties, and because it is the only to maintain the osteo-reparative, osteogenic, osteoinductive and osteoconductive properties, besides reducing the costs and providing more bone. The major disadvantage of this type of graft is the greater morbidity to the patient, besides the need for two surgeries. 5,6
Despite the wide possibility of intra- or extraoral graft donor sites, 6 selection of the donor area should consider the volume of bone loss and consequently the amount of bone required for reconstruction, the type of bone defect presented by the patient, the surgical-prosthetic planning, geometric conformation of the ridge, general conditions of the patient and good three-dimensional positioning of the anchorages to withstand the occlusal forces. 6 The iliac crest is the preferred donor site for large reconstructions. 5,6 Besides allowing easy surgical access due to its anatomical location, it provides a large amount of bone, both cortical and cancellous. In the present case, the iliac crest graft, together with a high-profile 2.0 system plate, proved to be a good treatment modality, without postoperative complications.
The biomodel was used to facilitate the surgical planning. Biomodels allow simulation of osteotomies and resection techniques, as well as complete planning of the most diverse types of oral and maxillofacial surgery. This tends to reduce the surgical time and consequently the period of anesthesia, as well as the risk of infection, with improved outcomes. 6
CONCLUDING REMARKS
Even though it is a rare lesion, the calcifying epithelial odontogenic tumor presents several treatment options. Among these, surgical resection and reconstruction with titanium plate and application of Carnoy solution, together with iliac crest graft and subsequent prosthetic rehabilitation with implants, proved to be an efficient treatment modality, since esthetic and functional results were satisfactorily obtained. The patient in the present case is under clinical follow-up, without signs of lesion relapse.
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Rydin K, Sjostrom M, Warfvinge G. Clear cell variant of intraosseous calcifying epithelial odontogenic tumor: a case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol. 2016;122(4):e125-30. Shetty SJ, Pereira T, Desai RS. Peripheral clear cell variant of calcifying epithelial odontogenic tumor: case report and review of the literature. Head Neck Pathol. 2016;10(4):481-5. Mariano RC, Oliveira MR, Silva AC, Ferreira DH, Almeida OP. Guided bone regeneration following surgical treatment of a rare variant of Pindborg tumor: a case report. J Oral Sci. 2014;56(1):95-8. Chen Y, Wang TT, Gao Y, Li TJ. A clinicopathologic study on calcifying epithelial odontogenic tumor: with special reference to Langerhans cell variant. Diagn Pathol. 2014 Feb 20;9:37. 5.
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8. Wadhwan V, Sharma P, Bansal V. A rare case of hybrid odontogenic tumor: Calcifying epithelial odontogenic tumor combined with ameloblastoma. J Oral Maxillofac Pathol. 2015 May-Aug;19(2):268. 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 Jan;12(1):78-84. Rani V, Masthan MK, Aravindha B, Leena S. Aggressive calcifying epithelial odontogenic tumor of the maxillary sinus with extraosseous oral mucosal involvement: a case report. Iran J Med Sci. 2016 Mar;41(2):145-9. Caliaperoumal SK, Gowri S, Dinakar J. Pindborg tumor. Contemp Clin Dent. 2016 Jan-Mar;7(1):95-7. 9. Priya S, Madanagopaal LR, Sarada V. Pigmented
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Maxillofac Pathol. 2016 Sept-Dec;20(3):548. 10. Costa Mendes L, Sauvigne T, Guiol J. [Morbidity of autologous bone harvesting in implantology: Literature review from 1990 to 2015]. Rev Stomatol Chirurg Maxillo-facial Chirurg Oral. 2016;117(6):388-402.
