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https://doi.org/10.15644/asc49/2/10

Cystic Shape Cemento-Ossifying Fibroma of Ethmoid Sinus

Dražen Shejbal ; OB Varaždin, Croatia
Gabrijela Vonsović ; Zupanja Health Centre, Croatia
Tomislav Baudoin ; School of Medicine University of Zagreb, ‘Sestre milosrdnice’ University Hospital, Zagreb, Croatia
Davor Vagić ; School of Dental Medicine, University of Zagreb; ‘Sestre milosrdnice’ University Hospital’, Zagreb, Croatia

Puni tekst: engleski, pdf (260 KB) str. 158-161 preuzimanja: 439* citiraj
APA 6th Edition
Shejbal, D., Vonsović, G., Baudoin, T. i Vagić, D. (2015). Cystic Shape Cemento-Ossifying Fibroma of Ethmoid Sinus. Acta stomatologica Croatica, 49 (2), 158-161. https://doi.org/10.15644/asc49/2/10
MLA 8th Edition
Shejbal, Dražen, et al. "Cystic Shape Cemento-Ossifying Fibroma of Ethmoid Sinus." Acta stomatologica Croatica, vol. 49, br. 2, 2015, str. 158-161. https://doi.org/10.15644/asc49/2/10. Citirano 18.09.2021.
Chicago 17th Edition
Shejbal, Dražen, Gabrijela Vonsović, Tomislav Baudoin i Davor Vagić. "Cystic Shape Cemento-Ossifying Fibroma of Ethmoid Sinus." Acta stomatologica Croatica 49, br. 2 (2015): 158-161. https://doi.org/10.15644/asc49/2/10
Harvard
Shejbal, D., et al. (2015). 'Cystic Shape Cemento-Ossifying Fibroma of Ethmoid Sinus', Acta stomatologica Croatica, 49(2), str. 158-161. https://doi.org/10.15644/asc49/2/10
Vancouver
Shejbal D, Vonsović G, Baudoin T, Vagić D. Cystic Shape Cemento-Ossifying Fibroma of Ethmoid Sinus. Acta stomatologica Croatica [Internet]. 2015 [pristupljeno 18.09.2021.];49(2):158-161. https://doi.org/10.15644/asc49/2/10
IEEE
D. Shejbal, G. Vonsović, T. Baudoin i D. Vagić, "Cystic Shape Cemento-Ossifying Fibroma of Ethmoid Sinus", Acta stomatologica Croatica, vol.49, br. 2, str. 158-161, 2015. [Online]. https://doi.org/10.15644/asc49/2/10
Puni tekst: hrvatski, pdf (260 KB) str. 158-161 preuzimanja: 200* citiraj
APA 6th Edition
Shejbal, D., Vonsović, G., Baudoin, T. i Vagić, D. (2015). Cistični oblik cemento-osificirajućeg fibroma etmoidnog sinusa. Acta stomatologica Croatica, 49 (2), 158-161. https://doi.org/10.15644/asc49/2/10
MLA 8th Edition
Shejbal, Dražen, et al. "Cistični oblik cemento-osificirajućeg fibroma etmoidnog sinusa." Acta stomatologica Croatica, vol. 49, br. 2, 2015, str. 158-161. https://doi.org/10.15644/asc49/2/10. Citirano 18.09.2021.
Chicago 17th Edition
Shejbal, Dražen, Gabrijela Vonsović, Tomislav Baudoin i Davor Vagić. "Cistični oblik cemento-osificirajućeg fibroma etmoidnog sinusa." Acta stomatologica Croatica 49, br. 2 (2015): 158-161. https://doi.org/10.15644/asc49/2/10
Harvard
Shejbal, D., et al. (2015). 'Cistični oblik cemento-osificirajućeg fibroma etmoidnog sinusa', Acta stomatologica Croatica, 49(2), str. 158-161. https://doi.org/10.15644/asc49/2/10
Vancouver
Shejbal D, Vonsović G, Baudoin T, Vagić D. Cistični oblik cemento-osificirajućeg fibroma etmoidnog sinusa. Acta stomatologica Croatica [Internet]. 2015 [pristupljeno 18.09.2021.];49(2):158-161. https://doi.org/10.15644/asc49/2/10
IEEE
D. Shejbal, G. Vonsović, T. Baudoin i D. Vagić, "Cistični oblik cemento-osificirajućeg fibroma etmoidnog sinusa", Acta stomatologica Croatica, vol.49, br. 2, str. 158-161, 2015. [Online]. https://doi.org/10.15644/asc49/2/10

Rad u XML formatu

Sažetak
Cemento-ossifying fibromas are a group of rarely occurring benign tumours, developing from the periodontal membrane and varying considerably in appearance and in the progress of the disease. Their common feature is higher or lower production of cemental tissue. In most cases the tumours are small because their cementoma mature quickly and become inactive, which causes the tumour to stop growing. They develop most frequently in the mandible and also in the maxilla. Other sites, such as paranasal cavities, soft tissues and bones of the head, are extremely rare. The case of a cemento-fibrosing tumour with psammoma infiltrations, developing from the ethmoid sinus in a nine-year-old girl is reported. Due to frontal headaches and sight defects as well as impaired vision on the right side, NMR was done, which showed a mucocele of the front and rear ethmoid with destruction of the orbital wall and a breakthrough into the orbit. The right maxillary sinus showed a visible retention and a thickened mucous membrane. A rhinoscopy revealed a ball-shaped spherical mass in the medial nasal meatus, which was defined as concha bullosa. An endoscopic examination showed that the tumour protruded in front of the medium nasal concha into the right nasal cavity, softened the ethmoid roof, penetrated toward the base of the skull, adhered and pushed the orbit. It was removed by FESS technique, and PHD revealed subsequently that it was not a mucocele but a cemento-ossifying fibroma.

Ključne riječi
Fibroma; Cementoma; Ethmoid Sinus; Mucous Membrane; Turbinates

Hrčak ID: 139979

URI
https://hrcak.srce.hr/139979

▼ Article Information



Introduction

Cementum is the bonelike, mineralised tissue covering the roots of vital teeth. It is a specialised connective tissue, similar to the osseous tissue, and the most important difference is cemental avascular structure. Cementoma develop from multipotent cells of the periodontal membrane, and by their multipotency those cells may produce connective tissue, cementum or bone. Pathological development of the periodontal membrane may thus result in the formation of tumours, composed of connective tissue, cementum and lamellar bone in different ratios (1).

Benign fibrous ossifying lesions are a problem in diagnostics and classification. The WHO classification from 1992 divides them into tumour lesions; cementifying fibroma and ossifying fibroma, and fibrous dysplasias, which are not tumours in the narrow sense. Numerous classifications aimed to classify tumours according to their clinical picture, aggressiveness and prognosis. The terms thus introduced, such as "juvenile active ossifying fibroma", "aggressive", "active", "psammomatoid", "juvenile active", are still rather controversial (2). Special clinical and diagnostic challenges are tumours located outside the jaws.

Case Report

We report a case of a nine-year-old girl, referred to our hospital because of frontal headaches, impaired vision and pain in the right eye. Her family's medical history was unremarkable; the patient is sensitive to dermatophagoides and pollen. All laboratory test results and lung x-rays were negative. The status at admission to hospital revealed a ball-shaped mass in the medium nasal meatus, which was defined as a concha bullosa. There was no pathological secretion; other ENT exams were normal.

MR imaging showed a mucocele of the front and rear ethmoid, destroying the orbital wall and breaking into the orbit, retention and thickened mucous membrane of the right maxillary sinus (Figure 1). The patient underwent an internal ethmoidectomy on the right side and extirpation of the mucocele by functional endoscopic surgery. Nasal endoscopy showed a mucocele in the medium nasal meatus, which was protruding fairly in front of the front pole of the medium nasal conchae. The formation was punctured and clear liquid was aspirated from the same. The mucocele wall peeled off from the ethmoid roof, the medium nasal shell and the periorbit to which it adhered and which was pushed by the mucocele. The endoscopic examination revealed softened bone of the base of the skull in the region of the posterior ethmoid. The natural orifice of the maxillary sinus dilated, in which thickened mucous membrane and some retention secretion were found. Endoscopies were repeated several times, without major mutilation consequences and without consequences for vision and nasal passages (Figure 2).

Figure 1 MR imaging before surgery; mucocele of the front and rear ethmoid, destructing the orbital wall and breaking into the orbit
ASC_49(2)_158-161-f1
Figure 2 MR imaging five years after surgery; without recurrence of disease
ASC_49(2)_158-161-f2

Discussion

It is possible to state that there are no uniform pathomorphological criteria for differentiation between cemento-ossifying dysplasias and cemento-ossifying fibroma. Histologically, fibromas are most often characterised by equal parts of calcified tissue and fibrous tissue; however, the ratio between these two tissues can vary, depending on the fibroma subtype. Calcified structures show a grid-like or an isolated lamellar structure with predominant osteoblasts and a few osteoclasts. Round or lobulated bone density mass may be more or less incorporated into the tumour, and their prevalence shows an image of a "cementifying fibroma". The presence of mineralised ossicles, "psammoma", is mentioned in recent literature. These are mineralised oval ossicles, not rimmed with osteoblasts. Connective tissue is composed of layers of spindle fibroblastic or stellate cells with focal areas of storiform patterns. Such microscopic composition is also directly responsible for major macropathological and clinical features of tumours, particularly in contrast to related dysplasias, and these are radiolucency, encapsulation, and slow and non-infiltrative growth (3, 4).

Most often, cementifying fibromas occur periapically in the maxilla and mandible; which is logical in view of its origin from the periodontal ligament. There are three clinical stages in their growth. The first stage destroys the bone, the tumour is without infiltrations of bone and cementum; in the second stage there are infiltrations of bone and cementum, and the tumour becomes opacifying luminiscent. When it reaches the third stage, the tumour stops to grow. It is marked by a well-demarcated, radiographically hyperlucent lesion and bone lesions. An average tumour diameter after the end of its growth is 3.8 cm (5).

Cementoma with sites outside the maxilla and mandible are quite different because they remain in an immature, active stage, which results in destructive growth and frequent relapses. The first case was described by Krausen et al. in 1977 (6). Tumour development outside the jaws is explained with ectopic cells of the periodontal membrane originating from the primitive mesenchymal ridge, which can appear there from embryologically underdeveloped tissue, after a surgery or after infection (7-9). Predominance of psammomatoid cells is found in approximately 11% of all cementifying osteofibroma. However, the frequency of cells increases with the distance between the site and the jaws. Thus psammoid cells would presuppose more aggressive behaviour of tumours with untypical sites: ethmoid and frontal sinuses, bones and soft tissues of the head (9).

Until the mid-1990s, the occurrence of cemento-ossifying fibromas outside the jaws was considered a rarity. However, in recent times, several cases are reported each year. The case reported above is extremely rare due to a cystic shape of the lesion itself. Fujimoto et al. reported first on such a case. Upon biochemical analysis of the aspirate they attempted to explain the cystic shape of the lesion with a rupture of the blood vessel and consequential compression necrosis of the tumour itself (10). Although aggressive, prone to relapses and penetration into surrounding vital structures of the face, cementifying fibromas outside the area of the jaws have a good prognosis, without mutilating consequences. Resection is the therapy of choice. Malignant alteration is possible, however, rarely (1, 4, 7, 10).

References

1 

Shanmugham MS. Cementifying fibroma of the ethmoidal sinus. J Laryngol Otol. 1984;98:639–42. DOI: http://dx.doi.org/10.1017/S0022215100147218 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/6736762

2 

Brannon RB, Fowler CB. Benign fibro osseous lesions: a review of current concepts. Adv Anat Pathol. 2001;8(3):126–43. DOI: http://dx.doi.org/10.1097/00125480-200105000-00002 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/11345237

3 

Su L, Weathers DR, Waldron CA. Distinguishing features of focal cemento-osseous dysplasia and cemento-ossifying fibromas. I. A pathologic spectrum of 316 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1997;84:301–9. DOI: http://dx.doi.org/10.1016/S1079-2104(97)90348-6 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/9377196

4 

Waldrom CA. Fibro-osseous lesions of the jaws. J Oral Maxillofac Surg. 1993;51:828–35. DOI: http://dx.doi.org/10.1016/S0278-2391(10)80097-7 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/8336219

5 

Krausen AS, Pullen PA, Zoografakis G, Cementoma II. Aggressive cemento-ossifying fibroma of the ethmoid region. Arch Otolaryngol. 1977;103:371–3. DOI: http://dx.doi.org/10.1001/archotol.1977.00780230093017 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/869775

6 

Su L, Weathers DR, Waldron CA. Distinguishing features of focal cemento-osseous dysplasia and cemento-ossifying fibromas. Clinical and radiologic spectrum of 316 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1997;84:540–9. DOI: http://dx.doi.org/10.1016/S1079-2104(97)90271-7 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/9394387

7 

Eversole LR, Leider AS, Nelson K. Ossifying fibroma: a clinicopathological study of sixty four cases. Oral Surg Oral Med Oral Pathol. 1985;60:510. DOI: http://dx.doi.org/10.1016/0030-4220(85)90239-7

8 

Cakir B, Karadayi N. Ossifying fibroma in the nasopharynx. A case report. Clin Imaging. 1991;15:290–2. DOI: http://dx.doi.org/10.1016/0899-7071(91)90122-C PubMed: http://www.ncbi.nlm.nih.gov/pubmed/1742681

9 

Jung LS, Choi KH, Park YA, Song CH, Kwon MS. Cemento-ossifyng fibroma presenting as a mass of the parapharyngeal and masticator space. AJNR Am J Neuroradiol. 1999;20:1744–6. PubMed: http://www.ncbi.nlm.nih.gov/pubmed/10543652

10 

Fujimoto Y, Katoh M, Miyata M, Kawai T, Saito K, Morita M. Cystic cemento ossifying fibroma of the ethmoidal cells (a case report). J Laryngol Otol. 1987;101(9):946–52. DOI: http://dx.doi.org/10.1017/S0022215100103032 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/3668378


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