Global Journal of Oral Science, 2017, 3, 33-36 33

E-ISSN-2414-2050 © 2017 Global Journal of Oral Science

Pindborg Tumor Involving Maxillary Sinus and Ethmoid Plate in Pediatric Patient: A Case Report

Andrea Tesei1, Marco Mascitti2,*, Daniele Gianfelici1, Alessandra Nori1, Andrea Santarelli2, and Vittorio Zavaglia1

1Special and Surgical Stomatology Department, “Ospedali Riuniti” Hospital of Ancona, Ancona, Italy and 2Department of Clinical Specialistic and Dental Sciences, Marche Polytechnic University, Ancona, Italy

Abstract: Calcifying epithelial odontogenic tumor (CEOT), also known as Pindborg tumor, is rare benign tumor deriving from odontogenic epithelium. The mean age at diagnosis is 40.3 years for the central variant and 31.8 years for the peripheral form. We report a case of 12 years-old female patient with maxillary and ethmoid sinus involvement treated with conservative surgical approach and Leukocyte and Platelet Rich Fibrin (LPRF) regenerative procedures.

Keywords: Odontogenic tumor, CEOT, Pindborg Tumor, Pediatric.

1. INTRODUCTION

Calcifying epithelial odontogenic tumor (CEOT), also known as Pindborg tumor, is a rare, benign but locally aggressive odontogenic tumor, described for the first time by Pindborg in 1955 [1]. CEOT represents less than 1% of all odontogenic neoplasms [2], and approxi- mately 200 cases have been reported until date.

The vast majority of cases occurs at intraosseous level, while peripheral CEOT accounts for less than 5% of cases [3]. Pediatric cases of odontogenic tumors are extremely rare; therefore, we want to describe an interesting case referred at Special and Surgical Stomatology Department, “Ospedali Riuniti” Hospital of Ancona: a 12-year-old girl with sinus localization of CEOT.

2. CASE REPORT

A 12-year-old female patient was reported to Com- plex Operating Unit of Special and Surgical Stomatol- ogy Department (“Ospedali Riuniti” Hospital of Ancona, Italy) for the evaluation of a worsening pain and swelling from upper left posterior region of maxilla for 3 months. No discharge and numbness were associated. Past medical history was insignificant.

Extraoral examination showed a diffuse swelling on the left side of the face with associated facial asym- metry. Palpation confirmed the findings, revealing a non-tender, non-fluctuant and hard swelling. The neck was soft with no evidence of lymphadenopathy or tenderness.

*Address correspondence to this author at the Department of Clinical Specialistic and Dental Sciences, Marche Polytechnic University, Ancona, Italy Via Tronto 10, 60126 Ancona, Italy; Phone +39-071-2206226; Fax +39-071-2206221; E-mail: marcomascitti86@hotmail.it

Intraoral examination showed swelling area extend- ing from 2.6 to 2.3, associated with 2.5 element agenesis and persistent 6.5 deciduous tooth. Vitality tests on near teeth reveled positive results and no alterations in color, shape and mobility were reported (Figure 1a).

Radiographic examination was conducted using Cone Beam Computed Tomography (CBCT), revealing a hypodense formation in the left jaw, rounded, with regular contours, of about 3.7 cm in diameter, with 2.5 tooth embedded. Margins of the lesion erode the root of the 6.5 element, the lower wall of the maxillary sinus and the lateral plate of the left ethmoid sinus (Figure 2).

Differential diagnosis included: residual cyst, denti- gerous cyst, keratocystic odontogenic tumor, calcifying epithelial odontogenic tumor, ameloblastoma (multi- cystic type), and odontogenic myxoma.

To reduce the risk of recurrence, radical surgical resection of the mass with clean margins was conduct- ed. Under general anesthesia, a mucoperiosteal flap was conducted in vestibular surface of left maxilla from 2.7 to 2.1, using piezoelectric bone surgery (Mectron Medical Technology, I-16042 Carasco). Osteotomy of vestibular plate in 2.5 position was performed and after exposition of the cleavage plane, the cystic lesion was enucleated (Figures 1b, 1c). A small fenestration was observed between residual cavity and maxillary sinus. The remaining cavity was filled with platelet-rich fibrin (PRF) (Figure 1d) and sutured using 3-0 silk suture (Figure 1e).

Gross pathology showed a white specimen, includ- ing a tooth, measuring 2.7 x 2 cm in size. The cut revealed calcifying areas. Histopathology showed a

34 Global Journal of Oral Science, 2017, Vol. 3 Tesei et al.

neoplasm with solid nests growth pattern, with intr- acytoplasmic lumen and ductal-like structure. A fibrous capsule enclosing epithelioid-like cells with abundant and eosinophilic cytoplasm. Nuclei were pleomorphic in appearance with rare mitosis.

Large areas of amorphous, eosinophilic, hyalinized material (amyloid-like) were observed and Liesegang ring appearance calcification (Figure 1f). Immuno- histochemical analysis revealed positivity for CK19, p63 and bcl2, while CK7 and CK20 were negative.

Figure 1: Diagnostic-therapeutic process: a) clinical aspect; b) exposition of the cystic lesion; c) enucleation of the cystic lesion; d) filling of remaining cavity using PRF; e) flap sutured; f) photomicrograph showing solid nests growth pattern with large areas of amorphous, eosinophilic, hyalinized material (hematoxylineosin-eosin, original magnification x40).

Pindborg Tumor Involving Maxillary Sinus and Ethmoid Plate Global Journal of Oral Science, 2017, Vol. 3, 35

Proliferative activity, evaluated with Mib-1, was 2%. The histopathological features suggested diagnosis of calcifying epithelial odontogenic tumor.

Post operation period was favorable for the patient; a follow-up CBCT was conducted 1 and 2 years after, revealing the formation of a new alveolar bridge (Figures 3a, 3b).

3. DISCUSSION

We have reported an extremely rare case of pediat- ric CEOT. This tumor, as in this case, frequently occurs at intraosseous level, while less than 5% of cases can develops within gingival tissues as a nodular mass. There is no sex prevalence, and the onset age range is from first to tenth decade, more frequently in the 20-60

Figure 2: CBCT performed before surgery: ethmoid and maxillary sinus involvement. a) lower margin; b) upper margin.

Figure 3: Follow-up CBCT: a) 1 year control; b) 2 years control, formation of alveolar bridge.

36 Global Journal of Oral Science, 2017, Vol. 3 Tesei et al.

years age group and with a peak of incidence in the fifth decade [4]. Posterior mandibular area is the most frequently involved region by CEOT; moreover, this tumor can be associated with an impacted, displaced tooth or root resorption due to the ability to infiltrate surrounding tissues [5].

The young age, the small dimensions and the presence of well-defined borders justify more conser- vative approaches, such as enucleation or curettage, followed by judicious removal of a thin layer of bone adjacent to the lesion.

Recurrence free follow-up of the patient make think that conservative approach is an optimal way to treat this tumor [6]. Lesions greater than 4 cm without well-defined radiological margins need to be treated with segmental resections and reconstruction procedures [7], in order to reduce the risk of recurrence, which is about 10-15% [8].

In our experience, PRF has proved to be an optimal strategy to encouraging and speed up healing as we can observe in follow-up CBCT. Several investigations should be conducted to develop a clear surgical protocol for CEOT improving use of piezoelectric bone surgery, a safe and accurate way to perform this treatment. Several studies describe its use in dental therapeutics but no one in CEOT treatment.

In conclusion, even if CEOT typically arises in adult patients, this case demonstrates that also in very young patient can be detected. The piezosurgery conservative

enucleation, associated to PRF can be considered the election treatment for limited size lesions.

4. STATEMENTS

This article does not contain any studies with human or animal subjects performed the authors.

Conflict of interest: none declared.

REFERENCES

[1] Pindborg JJ. Calcifying epithelial odontogenic tumour. Acta Pathol Microbiol Scand 1955; 111: 71.

[2] Angadi PV, Rekha K. Calcifying epithelial odontogenic tumor (Pindborg tumor). Head Neck Pathol 2011; 5: 137-9.

[3] Goode RK. Calcifying epithelial odontogenic tumor. Oral Maxillofac Surg Clin North Am 2004; 16: 323-31.

[4] Franklin CD, Pindborg JJ. The calcifying epithelial odontogenic tumor. A review and analysis of 113 cases. Oral Surg Oral Med Oral Pathol 1976; 42(6): 7 53-65.

[5] Patino B, Fernàndez-Alba J, Garcia-Rozado A, Martin R, Lòpez-Cedrùn JL, Sanromàn 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.

[6] Kim Y, Choi BE, Ko S-O. Conservative approach to recurrent calcifying cystic odontogenic tumor occupying the maxillary sinus: a case report. J Korean Assoc Oral Maxillofac Surg 2016; 42: 315.

[7] Califano L, Zupi A, Vetrani A, Fulciniti F, Giardino C. Calcifying odontogenic epithelial tumor or Pindborg’s tumor. Apropos of a case. Rev Stomatol Chir Maxillofac 1993; 94: 110-4.

[8] 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.

Received on 12-10-2017 Accepted on 23-10-2017 Published on 16-12-2017 © 2017 Tesei et al.; Licensee Global Journal of Oral Science. This is an open access article licensed under the terms of the Creative Commons Attribution Non-Commercial License (http: //creativecommons.org/licenses/by-nc/3.0/), which permits unrestricted, non-commercial use, distribution and reproduction in any medium, provided the work is properly cited.