glandular odontogenic cyst (sialo-odontogenic cyst)

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Glandular odontogenic cyst (sialo-odontogenic cyst) A case report G. Ficarra 1, L. Chou 2, E. Panzoni 1 1Division of Maxillofacial Surgery, University of Florence, Italy, and 2Division of Oral Pathology, University of California, San Francisco, USA G. Ficarra, L. Chou, E. Panzoni: Glandular odontogenic cyst (sialo-odontogenie cyst). A ease report. Int. J. Oral Maxillofac. Surg. 1990; 19: 331-333. Abstract. A case of a rare cyst of the jaw bones is reported. This lesion appears to be a new entity and has been named glandular odontogenic cyst (GOC) or sialo-odontogenic cyst. The clinical and histological features and the differential diagnosis are discussed. Key words: glandular odontogenic cyst; sialo- odontogenic cyst; cyst. Accepted for publication 21 June 1990 In a recent article, Gardner et al. 2 intro- duced the term glandular odontogenic (GOC) for a cyst that possibly rep- resents a new entity which presents with unusual histopathological features and tends to recur. This lesion occurs in either the mandible or maxilla and does not appear to show preference for age or gender and may become quite large. To our knowledge, 11 cases have been reported in the literature 2,3,4,5. The histopathological aspects include the presence of stratified epithelium of variable thickness, pools of mucicar- mine-positive material, interspersed mucous cells, eosinophilic cuboidal cells located on the superficial layer of the epithelium that give a papillary appear- ance, and epithelial cells arranged to form swirling areas 2. Cysts with similar characteristics have previously been re- ported under the name of sialo-odonto- genie cysts 4. The aim of this article is to describe a patient with a mandibular lesion ex- hibiting features of GOC. The clinical and histological features and the differ- ential diagnosis of this rare cyst are dis- cussed. lesion was multilocular and in some areas had a honeycomb appearance (Fig. 1). An incisional biopsy was performed. The lesion consisted of a thin-walled multilobular cyst, which was easily and partially euretted from the surrounding bone. The tissue was fixed in 10% neutral buf- fered formalin, embedded in paraffin, sec- tioned at 6 pm and stained with hematoxylin and eosin, and mucicarmine. Histological examination revealed a multicystic lesion. The cystic spaces were lined by nonkeratini- zed, stratified, squamous epithelium which varied in thickness. Lacunae present within the epithelial layer were replete with muein- positive material. Epithelial cells arranged to form ball-like structures were also noted (Fig. 2). Mucous and eosinophilic cuboidal cells were present in some areas of the lining epi- thelium giving a papillary appearance. Cilia were not dearly seen. Mierocysts, lined by secretory epithelium, appeared also to be en- trapped in the surrounding connective tissue (Fig. 3). These mierocysts were lined by eosinophilic cells and contained mucin-posi- five material. Irregularly-shaped calcifi- cations were seen in the connective tissue. At the time of the initial biopsy a diagnosis of a central mucoepidermoid tumor was made and the lesion was subsequently re- moved by a mandibular resection. Further consultations considered the diagnosis of "si- alo-odontogenic cyst", "botyroid odonto- genie cyst," "metastatic tumor" and "cyst of unknown origin." The patient underwent a bone graft after 8 months, and at present (34 months later) there are no signs of recurrence. Case report A 64-year-old edentulous woman was re- ferred because of a painful swelling in the anterior mandible. She reported that the lesion had been present for 1 year, and that her lower lip was slightly numb on both sides. Examination revealed a bony mass with buc- cal and lingual expansion which involved the midline portion of the mandible. The radio- graphs showed a large (4 x 10 cm), well-de- fined radiolucency in the symphysis. The Fig. 1. Large multilocular radiolucency of the anterior mandible. Note the honeycomb aspect.

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Glandular odontogenic cyst (sialo-odontogenic cyst) A case report

G. Ficarra 1, L. Chou 2, E. Panzoni 1 1Division of Maxillofacial Surgery, University of Florence, Italy, and 2Division of Oral Pathology, University of California, San Francisco, USA

G. Ficarra, L. Chou, E. Panzoni: Glandular odontogenic cyst (sialo-odontogenie cyst). A ease report. Int. J. Oral Maxillofac. Surg. 1990; 19: 331-333.

Abstract. A case of a rare cyst of the j aw bones is reported. This lesion appears to be a new enti ty and has been n a m e d g landular odon togen ic cyst (GOC) or s ia lo-odontogenic cyst. The clinical and histological features and the differential diagnosis are discussed.

Key words: glandular odontogenic cyst; sialo- odontogenic cyst; cyst.

Accepted for publication 21 June 1990

In a recent article, Ga rdne r et a l . 2 in t ro- duced the term glandular odon togen ic (GOC) for a cyst tha t possibly rep- resents a new enti ty which presents wi th unusua l h is topathologica l features and tends to recur. This lesion occurs in ei ther the mand ib le or maxil la and does no t appea r to show preference for age or gender and may become quite large. To our knowledge, 11 cases have been repor ted in the l i terature 2,3,4,5.

The h is topathologica l aspects include the presence of stratified epi thel ium of var iable thickness, pools of mucicar- mine-posi t ive material , interspersed mucous cells, eosinophil ic cuboidal cells located on the superficial layer of the epi thel ium tha t give a papi l lary appear- ance, a n d epithelial cells a r ranged to form swirling areas 2. Cysts with similar characteris t ics have previously been re- por ted unde r the name of s ia lo-odonto- genie cysts 4.

The a im of this article is to describe a pa t ien t with a mand ibu la r lesion ex- h ib i t ing features of GOC. The clinical and histological features and the differ- ent ial diagnosis o f this rare cyst are dis- cussed.

lesion was multilocular and in some areas had a honeycomb appearance (Fig. 1).

An incisional biopsy was performed. The lesion consisted of a thin-walled multilobular cyst, which was easily and partially euretted from the surrounding bone.

The tissue was fixed in 10% neutral buf- fered formalin, embedded in paraffin, sec- tioned at 6 pm and stained with hematoxylin and eosin, and mucicarmine. Histological examination revealed a multicystic lesion. The cystic spaces were lined by nonkeratini- zed, stratified, squamous epithelium which varied in thickness. Lacunae present within the epithelial layer were replete with muein- positive material. Epithelial cells arranged to form ball-like structures were also noted (Fig. 2). Mucous and eosinophilic cuboidal cells w e r e present in some areas of the lining epi-

thelium giving a papillary appearance. Cilia w e r e not dearly seen. Mierocysts, lined by secretory epithelium, appeared also to be en- trapped in the surrounding connective tissue (Fig. 3). These mierocysts were lined by eosinophilic cells and contained mucin-posi- five material. Irregularly-shaped calcifi- cations were seen in the connective tissue.

At the time of the initial biopsy a diagnosis of a central mucoepidermoid tumor was made and the lesion was subsequently re-

m o v e d by a mandibular resection. Further consultations considered the diagnosis of "si- alo-odontogenic cyst", "botyroid odonto- genie cyst," "metastatic tumor" and "cyst of unknown origin."

The patient underwent a bone graft after 8 months, and at present (34 months later) there are no signs of recurrence.

Case report

A 64-year-old edentulous woman was re- ferred because of a painful swelling in the anterior mandible. She reported that the lesion had been present for 1 year, and that her lower lip was slightly numb on both sides. Examination revealed a bony mass with buc- cal and lingual expansion which involved the midline portion of the mandible. The radio- graphs showed a large (4 x 10 cm), well-de- fined radiolucency in the symphysis. The Fig. 1. Large multilocular radiolucency of the anterior mandible. Note the honeycomb aspect.

332 Ficarra et al.

Fig. 2. The cyst epithelium consists of stratified squamous epithelium which contains numerous pools of mucin-positive material. Arrows point to the ball-like epithelial structures present in the lower part of the epithelium. (H & E, x 10)

Discussion

The histogenesis of this apparently new entity is controversial and has been sub- ject to debate. PADAYACHEE & VAN WVK 4 discussed the possibility of intra- bony salivary gland tissue being the ori- gin and suggested the term sialo-odon- togenic cyst. These authors also de- scribed the features of this lesion as a combination of both botyroid odonto- genic cyst and mucoepidermoid tumor.

GARDNER et al. 2 reported 8 cases, which they named GOC because of the presence of mucous cells and mucous- containing spaces in the lining epithe- lium. They considered these cysts as

"undoubtedly" odontogenic and based their conclusion upon exclusion of other possibilities and on the histopatholog- ical features.

The findings of GARDNER et al. 2 lend support to an odontogenic origin be- cause often ball-like epithelial structures are found as commonly seen in lateral periodontal cysts, adenomatoid odonto- genic tumor and, rarely, in ameloblas- toma and dentigerous cyst. They also noticed irregularly shaped calcifications that are frequently seen in odontogenic cysts. One of their cases appeared to be associated with ameloblastoma.

PADAYACHEE t~ VAN WYK 4 described 2 cases of GOC located in the anterior

Fig. 3. Microcy~ts, lined by secretory epithelium, are present in the surrounding connective tissue. (H & E, x 10)

mandible. The lesions appeared to be circumscribed, multilocular radio- lucencies and were multicystic. Histo- logically, pools of mucin, mucous and citiated cells and plaques and nodular thickening of the lining epithelium were seen. The lesion recurred in one pgtient after curettage.

GARDNER et a l l reported 2 cases of the maxilla and 6 of the mandible. All presented as unilocular or multilocular radiolucencies, often with very large di- mensions. Two patients had a recur- rence after 3 years and 3 1/2 years, re- spectively.

GOC may be considered as a variant of botyroid odontogenic cyst. In the lat- ter, mucous cells and epithelial thicken- ings are common. Microcysts and mu- cous cells in the cyst lining have been observed in cases of odontogenic cysts and botyroid odontogenic cysts 1. HEI- KINHEIMO et al. 3 described a case of a botyroid odontogenic cyst with multiple recurrences which had clinical and his- tological features similar to those of GOC.

GOC should also be differentiated from the central mucoepidermoid carci- noma, especially the low-grade type. The latter is composed of a mixture of epidermoid cells, mucous cells and cys- tic spaces lined by a single layer of mu- cin-producing cells. Intermediate types ofmucoepidermoid carcinoma are more cellular and form solid nests of cells, while high-grade types manifest intense anaplasia, hyperchrornatism and fre- quent mitoses.

The present case presents features that make it difficult to differentiate the lesion from central mucoepidermoid tu- mor. Two consultant pathologists made a diagnosis of low grade mucoepider- mold carcinoma.

Considering the biological behavior of GOC, it would seem that a somewhat more aggressive surgical removal, rather than simple curettage, should be considered. Careful enucleation and fix- ation with Carnoy's solution may be the treatment of choice 6.

References

1. BROWN RM. Metaplasia and degenera- tion in odontogenic cysts in man. J Oral Pathol 1972: 1: 45-9.

2. GARDNER DG, KESSLER HP~ MORENCY R, SCHAFFNER DL. The glandular odonto- genic cyst: an apparent entity. J Oral Pa- thol 1988: 17: 359-66.

3. HEIKINHEIMO K, HAPPONEN R-P, FORSELL K, KUUSm~aTO A, VmTATEN I. A boty-

Fig. 4. High-power magnification exhibiting a pool of mucin-positive material (*) lined by eosinophilic cells (arrows). (H & E, × 40)

Glandular odontogenic cyst 333

roid odontogenic cyst with multiple re- currences. Int J Oral Maxillofac Surg 1989: 18: 10-13.

4. PADAYACHEE A, VAN WYK CW. Two cys- tic lesions with features of both the boty- roid odontogenic cyst and the central mu- coepidermoid tumor: sialo-odontogenic cyst? J Oral Pathol 1987: 16: 499-504.

5. SADEGHI E, WELDON L, KWON R SAMPSON E. Mucoepidermoid odonto- genie cyst: a lesion with features of both the botyroid odontogenic cyst and central mucoepidermoid tumor; a variation or a new entity! Proceedings, American Acad- emy of Oral Pathology, 44th Annual Meeting, April 21-26. 1990, San Diego. Abstract #43.

6. STOELINGA PJW, BRONKHORST B. The in- cidence, multiple presentation and recur- rence of aggressive cysts of the jaws. J Cranio Maxillofac Surg 1988: 16:18445.

Address: Giuseppe Fiearra Institute of Odontology and Stomatology University of Florence Policlinieo di Careggi Viale Morgagni 50139 Florence Italy