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Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report
YBJOM-5541;
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Available online at www.sciencedirect.com
ScienceDirect British Journal of Oral and Maxillofacial Surgery xxx (2018) xxx–xxx
Short communication
Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report U. Güls¸en a,∗ , Ö. Dereci b , E.A. Güls¸en a a b
Bülent Ecevit University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Zonguldak, Turkey Eski¸sehir Osmangazi University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Eski¸sehir, Turkey
Accepted 13 November 2018
Abstract Conservative treatment of odontogenic tumours with decompression or marsupialisation is not common, but can be done successfully in those with a cystic pattern. We present a calcifying epithelial odontogenic tumour that was treated by tube decompression and subsequent enucleation. © 2018 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Calcifying epithelial odontogenic tumour; Decompression; Pindborg Tumour
Introduction
Case report
Calcifying epithelial odontogenic tumour is a relatively rare lesion that is seen predominantly in the mandible.1 It is characterised by secretion of an odontogenic amyloid protein into the stroma, and over time, this protein will gradually become calcified.1 Small tumours are managed conservatively with curettage and enucleation (with the removal of the thin layer of bone surrounding the lesion).2 however, advanced surgical approaches such as segmental and marginal resections are used in larger and more aggressive lesions.3 We report a case of a calcifying epithelial odontogenic tumour that was treated with decompression, with a saline cuff used as a tube for irrigation.
A 35-year-old man was referred to the clinic for a routine oral examination. Panoramic radiographs showed a multilocular, radiolucent lesion in the left molar region that involved the roots of all the molar teeth with mandibular cortical expansion (Fig. 1). His medical history was not helpful. The third molar was extracted and an incisional biopsy specimen taken under local anaesthesia (articaine hydrochloride 40 mg/ml, adrenaline hydrochloride 0.012 mg/ml). The length of a saline cuff (Fig. 2) was adjusted to fit the extraction socket and sutured to the surrounding gingiva and oral mucosa with 3/0 polyglactin 910 (Vycril, Ethicon) for immobilisation. Irrigation with a saline solution was started on postoperative day two and continued three times/day. Histological examination of the biopsy specimen showed a cystic cavity that was lined with squamous eosinophilic cells with wide cytoplasm and large nuclei and surrounded by a wall of connective tissue with no prominent inflammation. There was a distinct reduction in the size of the lesion at six months’ follow up (Fig. 3). Enucleation of the remaining lesion was done at the end of the sixth month, and tooth
∗ Corresponding author at: Eskis¸ehir Osmangazi University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Mes¸elik Campus, 26480 Eskis¸ehir, Turkey. Tel.: no:+902222391303; Fax no:+902222391273. E-mail addresses: [email protected], [email protected] (U. Güls¸en).
https://doi.org/10.1016/j.bjoms.2018.11.008 0266-4356/© 2018 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Güls¸en U, et al. Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report. Br J Oral Maxillofac Surg (2018), https://doi.org/10.1016/j.bjoms.2018.11.008
YBJOM-5541; 2
No. of Pages 3
ARTICLE IN PRESS U. Gül¸sen et al. / British Journal of Oral and Maxillofacial Surgery xxx (2018) xxx–xxx
Fig. 1. Multilocular cystic lesion that had caused cortical expansion on the left posterior molar region and involved the roots of molar teeth. Fig. 4. Histopathological examination showed solid cell islands that had been formed by polygonal squamous cells with eosinophilic cytoplasm and large nuclei. There was an amyloid matrix, which was stained pink with crystal violet stain in the extracellular matrix. Mineralisation zones were seen in the amyloid matrix (haematoxylin and eosin, original magnification x 200).
Fig. 5. Complete healing at 13 months’ follow up after enucleation.
Fig. 2. Saline cuff was cut to adapt to the extraction socket and used for irrigation.
duction and calcifications. (Fig. 4) We made a final diagnosis of calcifying epithelial odontogenic tumour, which had healed completely (after enucleation) at 13 months’ followup (Fig. 5).
Discussion
Fig. 3. There was prominent regeneration of bone with complete disappearance of the sclerosis on the borders six months after decompression.
37 extracted under local anaesthesia (articaine hydrochloride 40 mg/ml, adrenaline hydrochloride 0.012 mg/ml). Histological examination showed islands of squamous neoplastic epithelial cells with homogeneous collagenous matrix pro-
Cystic epithelium may be the primary characteristic and main histological component of a calcifying epithelial odontogenic tumour, which presents as a unilocular cystic lesion on radiography.4 Azevedo et al5 reported that three of their 19 cases showed cystic epithelium associated with the solid tumour. It is debatable whether the growth of the lesion shows a cystic pattern, or if cystic degeneration occurs in the tumour when the proliferation reaches a peak level.4 In our patient there was a cystic pattern, which was histopathologically diagnosed at the time of incisional biopsy examination, and it encouraged the clinicians to decompress the cystic lesion. There was no response to decompression after six months and the remaining small amount of the tumour was enucleated. Complete evaluation of the enucleated lesion showed a solid
Please cite this article in press as: Güls¸en U, et al. Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report. Br J Oral Maxillofac Surg (2018), https://doi.org/10.1016/j.bjoms.2018.11.008
YBJOM-5541;
ARTICLE IN PRESS
No. of Pages 3
U. Gül¸sen et al. / British Journal of Oral and Maxillofacial Surgery xxx (2018) xxx–xxx
epithelial tumour that involved the production of amyloids and led to calcification. Conservative treatment of odontogenic tumours with decompression or marsupialisation is known to be particularly suitable for those tumours that include or show cystic components such as keratocystic odontogenic tumour, adenomatoid odontogenic tumour, calcifying odontogenic cyst, and unicystic ameloblastoma.6–8 Shakib et al9 reported that nasopharyngeal airways can be reliably used in the decompression and marsupialisation of unicystic ameloblastoma. In our case a more resilient saline cuff was used to establish a sound adaptation in the extraction socket. Xavier et al10 suggested that treatment of a mural unicystic ameloblastoma is possible with decompression and irrigation with 0.12% clorhexidine digluconate twice a day followed by enucleation. Emam et al8 reported a patient with a calcifying odontogenic cyst who had a two-stage treatment of decompression and enucleation that was similar to ours. Decompression is a conservative procedure that evacuates the hydrostatic pressure in the bony cavity of the lesion and induces regeneration of bone on the borders. It is simple and helps the surgeon to avoid neurovascular structures that are likely to be damaged with a more invasive approach. Odontogenic tumours may present cystic compartments within a solid form or show a completely cystic pattern without a solid component. The decompression of the cystic counterpart of a solid tumour, as described here, may be beneficial to reduce the size of the cyst and prepare the patient for a less invasive procedure. To the best of our knowledge, this is the first report of a conservative treatment of tube decompression followed by enucleation for a calcifying epithelial odontogenic tumour.
3
Ethics statement/confirmation of patient’s permisson No ethics approval was needed.The patient’s written permission was obtained. References 1. Wright JM, Deviliers P, et al. Calcifying epithelial odontogenic tumor. In: El-Naggar AK, Chan JKC, editors. WHO classification of head and neck tumours. 4th ed. IARC Press; 2017. p. 220–1. 2. Chomette G, Auriol M, Guilbert F. Histoenzymological and ultrastructural study of a bifocal calcifying epithelial odontogenic tumor. Characteristics of epithelial cells and histogenesis of amyloid-like material. Virchows Arch A Pathol Anat Histopathol 1984;403:67–76. 3. Singh N, Sahai S, Singh S, et al. Calcifying epithelial odontogenic tumor (Pindborg tumor). Natl J Maxillofac Surg 2011;2:225–7. 4. Gopalakrishnan R, Simonton S, Rohrer MD, et al. Cystic variant of calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:773–7. 5. Azevedo RS, Mosqueda-Taylor A, Carlos R, et al. Calcifying epithelial odontogenic tumor (CEOT): a clinicopathologic and immunohistochemical study and comparison with dental follicles containing CEOT-like areas. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:759–68. 6. Erdur EA, Ileri Z, Ugurluoglu C, et al. Eruption of an impacted canine in an adenomatid odontogenic tumor treated with combined orthodontic and surgical therapy. Am J Orthod Dentofacial Orthop 2016;149:923–7. 7. Dolanmaz D, Etoz OA, Pampu A, et al. Marsupialization of unicystic ameloblastoma: a conservative approach for aggressive odontogenic tumors. Indian J Dent Res 2011;22:709–12. 8. Emam HA, Smith J, Briody A, et al. Tube decompression for staged treatment of a calcifying odontogenic cyst — a case report. J Oral Maxillofac Surg 2017;75:1915–20. 9. Shakib K, Heliotis M, Gilhooly M. The nasopharyngeal airway: reliable and effective tool for marsupialisation. Br J Oral Maxillofac Surg 2010;48:386–7. 10. Xavier SP, de Mello-Filho FV, Rodrigues WC, et al. Conservative approach: using decompression procedure for management of a large unicystic ameloblastoma of the mandible. J Craniofac Surg 2014;25:1012–4.
Conflict of interest We have no conflicts of interest.
Please cite this article in press as: Güls¸en U, et al. Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report. Br J Oral Maxillofac Surg (2018), https://doi.org/10.1016/j.bjoms.2018.11.008
ARTICLE IN PRESS
No. of Pages 3
Available online at www.sciencedirect.com
ScienceDirect British Journal of Oral and Maxillofacial Surgery xxx (2018) xxx–xxx
Short communication
Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report U. Güls¸en a,∗ , Ö. Dereci b , E.A. Güls¸en a a b
Bülent Ecevit University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Zonguldak, Turkey Eski¸sehir Osmangazi University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Eski¸sehir, Turkey
Accepted 13 November 2018
Abstract Conservative treatment of odontogenic tumours with decompression or marsupialisation is not common, but can be done successfully in those with a cystic pattern. We present a calcifying epithelial odontogenic tumour that was treated by tube decompression and subsequent enucleation. © 2018 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Calcifying epithelial odontogenic tumour; Decompression; Pindborg Tumour
Introduction
Case report
Calcifying epithelial odontogenic tumour is a relatively rare lesion that is seen predominantly in the mandible.1 It is characterised by secretion of an odontogenic amyloid protein into the stroma, and over time, this protein will gradually become calcified.1 Small tumours are managed conservatively with curettage and enucleation (with the removal of the thin layer of bone surrounding the lesion).2 however, advanced surgical approaches such as segmental and marginal resections are used in larger and more aggressive lesions.3 We report a case of a calcifying epithelial odontogenic tumour that was treated with decompression, with a saline cuff used as a tube for irrigation.
A 35-year-old man was referred to the clinic for a routine oral examination. Panoramic radiographs showed a multilocular, radiolucent lesion in the left molar region that involved the roots of all the molar teeth with mandibular cortical expansion (Fig. 1). His medical history was not helpful. The third molar was extracted and an incisional biopsy specimen taken under local anaesthesia (articaine hydrochloride 40 mg/ml, adrenaline hydrochloride 0.012 mg/ml). The length of a saline cuff (Fig. 2) was adjusted to fit the extraction socket and sutured to the surrounding gingiva and oral mucosa with 3/0 polyglactin 910 (Vycril, Ethicon) for immobilisation. Irrigation with a saline solution was started on postoperative day two and continued three times/day. Histological examination of the biopsy specimen showed a cystic cavity that was lined with squamous eosinophilic cells with wide cytoplasm and large nuclei and surrounded by a wall of connective tissue with no prominent inflammation. There was a distinct reduction in the size of the lesion at six months’ follow up (Fig. 3). Enucleation of the remaining lesion was done at the end of the sixth month, and tooth
∗ Corresponding author at: Eskis¸ehir Osmangazi University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Mes¸elik Campus, 26480 Eskis¸ehir, Turkey. Tel.: no:+902222391303; Fax no:+902222391273. E-mail addresses: [email protected], [email protected] (U. Güls¸en).
https://doi.org/10.1016/j.bjoms.2018.11.008 0266-4356/© 2018 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Güls¸en U, et al. Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report. Br J Oral Maxillofac Surg (2018), https://doi.org/10.1016/j.bjoms.2018.11.008
YBJOM-5541; 2
No. of Pages 3
ARTICLE IN PRESS U. Gül¸sen et al. / British Journal of Oral and Maxillofacial Surgery xxx (2018) xxx–xxx
Fig. 1. Multilocular cystic lesion that had caused cortical expansion on the left posterior molar region and involved the roots of molar teeth. Fig. 4. Histopathological examination showed solid cell islands that had been formed by polygonal squamous cells with eosinophilic cytoplasm and large nuclei. There was an amyloid matrix, which was stained pink with crystal violet stain in the extracellular matrix. Mineralisation zones were seen in the amyloid matrix (haematoxylin and eosin, original magnification x 200).
Fig. 5. Complete healing at 13 months’ follow up after enucleation.
Fig. 2. Saline cuff was cut to adapt to the extraction socket and used for irrigation.
duction and calcifications. (Fig. 4) We made a final diagnosis of calcifying epithelial odontogenic tumour, which had healed completely (after enucleation) at 13 months’ followup (Fig. 5).
Discussion
Fig. 3. There was prominent regeneration of bone with complete disappearance of the sclerosis on the borders six months after decompression.
37 extracted under local anaesthesia (articaine hydrochloride 40 mg/ml, adrenaline hydrochloride 0.012 mg/ml). Histological examination showed islands of squamous neoplastic epithelial cells with homogeneous collagenous matrix pro-
Cystic epithelium may be the primary characteristic and main histological component of a calcifying epithelial odontogenic tumour, which presents as a unilocular cystic lesion on radiography.4 Azevedo et al5 reported that three of their 19 cases showed cystic epithelium associated with the solid tumour. It is debatable whether the growth of the lesion shows a cystic pattern, or if cystic degeneration occurs in the tumour when the proliferation reaches a peak level.4 In our patient there was a cystic pattern, which was histopathologically diagnosed at the time of incisional biopsy examination, and it encouraged the clinicians to decompress the cystic lesion. There was no response to decompression after six months and the remaining small amount of the tumour was enucleated. Complete evaluation of the enucleated lesion showed a solid
Please cite this article in press as: Güls¸en U, et al. Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report. Br J Oral Maxillofac Surg (2018), https://doi.org/10.1016/j.bjoms.2018.11.008
YBJOM-5541;
ARTICLE IN PRESS
No. of Pages 3
U. Gül¸sen et al. / British Journal of Oral and Maxillofacial Surgery xxx (2018) xxx–xxx
epithelial tumour that involved the production of amyloids and led to calcification. Conservative treatment of odontogenic tumours with decompression or marsupialisation is known to be particularly suitable for those tumours that include or show cystic components such as keratocystic odontogenic tumour, adenomatoid odontogenic tumour, calcifying odontogenic cyst, and unicystic ameloblastoma.6–8 Shakib et al9 reported that nasopharyngeal airways can be reliably used in the decompression and marsupialisation of unicystic ameloblastoma. In our case a more resilient saline cuff was used to establish a sound adaptation in the extraction socket. Xavier et al10 suggested that treatment of a mural unicystic ameloblastoma is possible with decompression and irrigation with 0.12% clorhexidine digluconate twice a day followed by enucleation. Emam et al8 reported a patient with a calcifying odontogenic cyst who had a two-stage treatment of decompression and enucleation that was similar to ours. Decompression is a conservative procedure that evacuates the hydrostatic pressure in the bony cavity of the lesion and induces regeneration of bone on the borders. It is simple and helps the surgeon to avoid neurovascular structures that are likely to be damaged with a more invasive approach. Odontogenic tumours may present cystic compartments within a solid form or show a completely cystic pattern without a solid component. The decompression of the cystic counterpart of a solid tumour, as described here, may be beneficial to reduce the size of the cyst and prepare the patient for a less invasive procedure. To the best of our knowledge, this is the first report of a conservative treatment of tube decompression followed by enucleation for a calcifying epithelial odontogenic tumour.
3
Ethics statement/confirmation of patient’s permisson No ethics approval was needed.The patient’s written permission was obtained. References 1. Wright JM, Deviliers P, et al. Calcifying epithelial odontogenic tumor. In: El-Naggar AK, Chan JKC, editors. WHO classification of head and neck tumours. 4th ed. IARC Press; 2017. p. 220–1. 2. Chomette G, Auriol M, Guilbert F. Histoenzymological and ultrastructural study of a bifocal calcifying epithelial odontogenic tumor. Characteristics of epithelial cells and histogenesis of amyloid-like material. Virchows Arch A Pathol Anat Histopathol 1984;403:67–76. 3. Singh N, Sahai S, Singh S, et al. Calcifying epithelial odontogenic tumor (Pindborg tumor). Natl J Maxillofac Surg 2011;2:225–7. 4. Gopalakrishnan R, Simonton S, Rohrer MD, et al. Cystic variant of calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:773–7. 5. Azevedo RS, Mosqueda-Taylor A, Carlos R, et al. Calcifying epithelial odontogenic tumor (CEOT): a clinicopathologic and immunohistochemical study and comparison with dental follicles containing CEOT-like areas. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:759–68. 6. Erdur EA, Ileri Z, Ugurluoglu C, et al. Eruption of an impacted canine in an adenomatid odontogenic tumor treated with combined orthodontic and surgical therapy. Am J Orthod Dentofacial Orthop 2016;149:923–7. 7. Dolanmaz D, Etoz OA, Pampu A, et al. Marsupialization of unicystic ameloblastoma: a conservative approach for aggressive odontogenic tumors. Indian J Dent Res 2011;22:709–12. 8. Emam HA, Smith J, Briody A, et al. Tube decompression for staged treatment of a calcifying odontogenic cyst — a case report. J Oral Maxillofac Surg 2017;75:1915–20. 9. Shakib K, Heliotis M, Gilhooly M. The nasopharyngeal airway: reliable and effective tool for marsupialisation. Br J Oral Maxillofac Surg 2010;48:386–7. 10. Xavier SP, de Mello-Filho FV, Rodrigues WC, et al. Conservative approach: using decompression procedure for management of a large unicystic ameloblastoma of the mandible. J Craniofac Surg 2014;25:1012–4.
Conflict of interest We have no conflicts of interest.
Please cite this article in press as: Güls¸en U, et al. Treatment of a calcifying epithelial odontogenic tumour with tube decompression: a case report. Br J Oral Maxillofac Surg (2018), https://doi.org/10.1016/j.bjoms.2018.11.008