- Email: [email protected]
Glandular odontogenic cyst: Clinicopathologic analysis of three cases
Glandular odontogenic cyst: Clinicopathologic analysis of three cases Mercedes Patron, MD,a Cesar Colmenero, MD.b and Javier Larrauri, UNIVERSIDAD
AUTONOMA
AND HOSPITAL
MD,c Madrid,
Spain
LA PAZ
The glandular odontogenic cyst is a rare cyst of odontogenic origin, first described in 1988 by Gardner et al. Three previously unreported glandular odontogenic cysts are presented; none recurred after the initial surgical treatment, and one example was associated with a squamous odontogenic tumor-like proliferation in the wall. Ten similar cases were found in the literature. and their clinical and roentgenographic features, and follow-up, have been compared with the present cases. (ORAL SURC ORAL MED ORAL PATHOL 1991;72:71-4)
T
he glandular odontogenic cyst (GOC) is an uncommon odontogenic cyst, originally described in 1988 by Gardner et al.’ as a distinct entity. In 1987 Padayacheeand Van Wyk2 reported two multilocular mandibular lesions with features of both botryoid odontogenic cyst and mucoepidermoid tumor, which they termed sialo-odontogenic cyst, and which appears similar to the case reported here. We report three additional casesof GOC, none of which recurred 20, 14, and 10 years after the initial surgical removal, respectively. One was associated with squamousodontogenic tumor-like proliferations in the wall, similar to those described by Wright3 in the walls of other odontogenic cysts. MATERIAL
Fig. 1. Case 2. Panoramic radiograph showing multilocular cyst of mandible.
AND METHODS
The clinical, radiographic, and histopathologic findings of three examples of GOC were reviewed. Tissues for light microscopy were fixed for 24 hours in 10% formalin saline solution and routinely processedfor paraffin sectioning. The 5 pm sections were stained with hematoxylin and eosin and periodic acid-Schiff, with and without digestion of glycogen. CASEREPORTS Case
1
A 45year-old man was seen in December 1969 with a tender swelling in the mandibular symphysis for 20 years,
Fig. 2. Case 2. Postoperative panoramic radiograph. Enucleation and endodontic surgery were performed 7 years previously.
aAssociate Professor, Department of Pathology, School of Medi-
cine, Universidad Autonoma, Hospital La Paz. bResident in Maxillofacial Surgery, Department of Surgery, Hos-
pital La Paz. cTitular Professor,Department of Pathology, School of Medicine, Universidad Autonoma, Hospital La Paz. 7/14/26330
that had increased its rate of growth in the last month. Physical examination revealed a soft, tender, 4 X 1 cm mass, with buccal expansion of the mandible. Radiologically, a unilocular radiolucency with broad sclerotic walls extending from the left mandibular central incisor to the right 71
72
Patron, Colmenero, and Larrauri
Fig. 3. Case 3. Occlusal and panoramic radiographs show unilocular radiolucency extending from lateral incisor to first molar and producing divergence of roots of teeth.
4. Photomicrograph shows that cyst is lined by multilayered squamous epithelium. (Hematoxylin-eosin stain; original magnification, X 10.) Fig.
ORAL SURC ORAL
MED ORAL PATHOL July 1991
Fig. 6. Mucous cells forming glands in somecases.(Hematoxylin-eosin stain; original magnification, X40.)
Fig. 7. Case 3. Photomicrograph shows cilia in surface cells and vacuolated basal cells. (Hematoxylin-eosin stain; original magnification, X63.)
second premolar with root resorption was observed. The presumptive clinical diagnosis was ameloblastoma, and, with the patient under general anesthesia,all affected teeth were extracted and a marginal mandibulectomy preserving the inferior border of the mandible was performed. The patient has been followed clinically and radiologically without recurrence for 20 years. Case 2
Fig. 5. Pools of mucus within spinous cell layer and cuboidal cells on surface of epithelium. (Hematoxylin-eosin stain; original magnification, X63.)
A 39-year-old man was seen at the outpatient clinic in 1979 with a left mandibular deformity associated with an intraoral swelling that had been increasing slowly. No pain or suppuration were elicited. Clinically, a massfrom the left canine to the right first molar with expansion of the buccal cortical plate was observed. Radiologically, a multilocular radiolucency from the left second premolar to and including the right first molar was seen. Root resorption was observed (Fig. 1). With the pa-
Glandular
Volume 72 Number I
73
Fig. 9. Case 1. Squamous odontogenic tumor-like epithelium without peripheral palisading. (Hematoxylin-eosin stain; original magnification, X25.)
Fig. 8. Case 3. Epithelial spheres are present in epithehum. (Hematoxylin-eosin stain; original magnification, x63.)
Table
odontogenic cyst
I. Reports of 13 casesof GOC
Case No. (reference)
Localization and x-ray jkdings
Treatment
I
I
Follow-up
1 (2)
69/M
Multilocular radiolucency of anterior mandible
Limited local excision
2 (2)
71/F
Limited local excision
3 (1)
21/F
Multilocular radiolucency of anterior mandible Maxilla, radiolucency
Recurrence of 3 yr; reexcision, no evidence of disease at 18 mo NA
Enucleation and endodontic
No recurrence after 3% yr
4 (1)
59/M
5 (1)
44/F
6 (1)
85/F
7 (1)
59/M
8 (1)
44/M
9 (1) 10 (1) 11
19/M 48/M 45/M
12
52/M
13
39/M
surgery Anterior mandible, unilocular radiolucency Anterior mandible, unilocular radiolucency Mandible, multilocular radiolucency Anterior mandible, unilocular radiolucency Maxilla, unilocular radiolucency Mandible, radiolucency Mandible, radiolucency Anterior mandible, unilocular radiolucency with sclerotic borders Maxilla, well-defined radiolucency Anterior mandible, multilocular radiolucency
Enucleation and endodontic surgery Enucleation NA Curettage Enucleation 9 yr after diagnosis Enucleation Enucleation Marginal mandibulectomy preserving inferior border Maxillectomy Enucleation, endodontics surgery
Recurrence at 3 yr 8 mo; enucleation Recurrence at 3 yr 3 mo; curettage NA No recurrence for 6 mo; lost to follow-up No recurrence in 2 yr No recurrence No recurrence No evidence of disease for 20 yr No evidence of disease for 14 yr No evidence of disease for 10 yr
NA. Not available.
tient under general anesthesia,the cyst was enucleated and root canal procedureswere performed from the right second premolar to the left second premolar (Fig. 2). The patient has beenfollowed without evidenceof disease for 10 years.
Case 3
A 52-year-old man was seen in 1968 for investigation of a gingival tumor. He had gingival swelling and facial asymmetry associatedwith a dull ache over the left anterior maxilla for 6 years. Radiologically, a unilocular radiolu-
74
Patron, Colmenero, and Larrauri
cency was identified, spanning the left lateral incisor to the left first molar without root resorption (Fig. 3). With the presumptive diagnosis of ameloblastoma, a partial maxillectomy was performed. The patient was followed until 1982 without any sign of recurrence. HISTOLOGIC
FINDINGS
The three lesions had the histomorphologic characteristics of the GOC as described by Gardner et al. 1 All lesions had a multilocular gross appearance and showed polycystic features microscopically. Histopathologic examination of the lesions revealed a stratified squamousepithelial lining of varying thickness, with a flat interface with the subjacent stroma (Fig. 4). The connective tissue wall in all caseswas relatively free of inflammatory cells. The superficial layer of the epithelium showed eosinophilic cuboidal and ciliated cells. Pools of periodic acid-schiffpositive, diastase-resistant material were present within the epithelium. Sometimes these mucus pools were lined by eosinophilic cuboidal cells, similar to those found in the lining epithelium (Fig. 5). Cases2 and 3 showed abundant mucous cells on the lining surface and sometimes formed mucous glands (Fig. 6). Occasionally the basal cells were vacuolated (Fig. 7). In case 3 the epithelial cells were arranged into spherical structures (Fig. 8). Case 1 presented islands of squamous epithelium that lacked peripheral palisading and that were in continuity with the cystic epithelial lining (Fig. 9). These squamous epithelial islands were completely confined to the cyst wall. None of our casesshowed calcifications or areas of hyalinization. DISCUSSION
The GOCs presented herein fulfill the diagnostic criteria proposedby Gardner et al.’ From the limited number of casesof GOC that have appeared in the literature (Table I), it seemsthat these lesions have a predilection for men (9/ 13) and for the mandible (lo/ 13). The age range is 19 to 85 years, with a mean age of 50 years. Radiographically the lesions are well defined, with a unilocular or multilocular pattern but without specific features that permit separation from other entities.
ORAL SURGORAL MED ORAL PATHOL July 1991
Of the 13 casesreviewed, 10 were adequately followed up and 3 recurred. The biologic nature of GOC seemsto be intermediate between odontogenic keratocyst and other cysts in size and multilocularity. Enucleation seemsto be adequate treatment despite the few reported cases. The presence of epithelial spheres and squamous odontogenic epithelial proliferations, which have been described in other odontogenic cysts, lends support to the speculation that GOC is odontogenic in nature. We agree with Gardner et al.,’ who suggest that the GOC is a histologic variant of the botryoid odontogenit cyst. The following features are in keeping with a botryoid odontogenic cyst: the occurrence during the fifth to the seventh decadesof life, the intrabody location in the mandibular premolar-canine region, the multilocular radiographic appearance,the recurrence of somelesions, and the presencehistologically of epithelial spheres. Recently Heikinheimo et a1.4found mucous cells in the biopsy specimen of a case of botryoid odontogenic cyst that recurred several times. The relationship, if any, of these two cysts should become clearer as more examples of each are studied.
REFERENCES Gardner DG, Kessler HP, Morency R, Schaffner DL. The glandular odontogenic cyst: an apparent entity. J Oral Pathol 1988;17:359-66. PadayacheeA, Van Wyk CW. Two cystic lesionswith features of both the botryoid odontogenic cyst and the central mucoepidermoid tumour: sialoodontogenic cyst. J Oral Pathol 1987;16:499-504. Wright JM. Squamous odontogenic tumor-like proliferations in odontogenic cysts. ORAL SURGORAL MED ORAL PATHOL 1919;47:354-8.
Heikinheimo K, Happonen RP, Forssell A, Kuusilehto A, Virtamen P. A botryoid odontogenic cyst with multiple recurrences. lnt J Oral Maxillofac Surg 1989;18:10-3.
Reprint requests:
Mercedes Patron, MD Departamento de Anatomia Patologica Hospital La Paz PaseoCastellana 26 1 28046 Madrid, Spain
AUTONOMA
AND HOSPITAL
MD,c Madrid,
Spain
LA PAZ
The glandular odontogenic cyst is a rare cyst of odontogenic origin, first described in 1988 by Gardner et al. Three previously unreported glandular odontogenic cysts are presented; none recurred after the initial surgical treatment, and one example was associated with a squamous odontogenic tumor-like proliferation in the wall. Ten similar cases were found in the literature. and their clinical and roentgenographic features, and follow-up, have been compared with the present cases. (ORAL SURC ORAL MED ORAL PATHOL 1991;72:71-4)
T
he glandular odontogenic cyst (GOC) is an uncommon odontogenic cyst, originally described in 1988 by Gardner et al.’ as a distinct entity. In 1987 Padayacheeand Van Wyk2 reported two multilocular mandibular lesions with features of both botryoid odontogenic cyst and mucoepidermoid tumor, which they termed sialo-odontogenic cyst, and which appears similar to the case reported here. We report three additional casesof GOC, none of which recurred 20, 14, and 10 years after the initial surgical removal, respectively. One was associated with squamousodontogenic tumor-like proliferations in the wall, similar to those described by Wright3 in the walls of other odontogenic cysts. MATERIAL
Fig. 1. Case 2. Panoramic radiograph showing multilocular cyst of mandible.
AND METHODS
The clinical, radiographic, and histopathologic findings of three examples of GOC were reviewed. Tissues for light microscopy were fixed for 24 hours in 10% formalin saline solution and routinely processedfor paraffin sectioning. The 5 pm sections were stained with hematoxylin and eosin and periodic acid-Schiff, with and without digestion of glycogen. CASEREPORTS Case
1
A 45year-old man was seen in December 1969 with a tender swelling in the mandibular symphysis for 20 years,
Fig. 2. Case 2. Postoperative panoramic radiograph. Enucleation and endodontic surgery were performed 7 years previously.
aAssociate Professor, Department of Pathology, School of Medi-
cine, Universidad Autonoma, Hospital La Paz. bResident in Maxillofacial Surgery, Department of Surgery, Hos-
pital La Paz. cTitular Professor,Department of Pathology, School of Medicine, Universidad Autonoma, Hospital La Paz. 7/14/26330
that had increased its rate of growth in the last month. Physical examination revealed a soft, tender, 4 X 1 cm mass, with buccal expansion of the mandible. Radiologically, a unilocular radiolucency with broad sclerotic walls extending from the left mandibular central incisor to the right 71
72
Patron, Colmenero, and Larrauri
Fig. 3. Case 3. Occlusal and panoramic radiographs show unilocular radiolucency extending from lateral incisor to first molar and producing divergence of roots of teeth.
4. Photomicrograph shows that cyst is lined by multilayered squamous epithelium. (Hematoxylin-eosin stain; original magnification, X 10.) Fig.
ORAL SURC ORAL
MED ORAL PATHOL July 1991
Fig. 6. Mucous cells forming glands in somecases.(Hematoxylin-eosin stain; original magnification, X40.)
Fig. 7. Case 3. Photomicrograph shows cilia in surface cells and vacuolated basal cells. (Hematoxylin-eosin stain; original magnification, X63.)
second premolar with root resorption was observed. The presumptive clinical diagnosis was ameloblastoma, and, with the patient under general anesthesia,all affected teeth were extracted and a marginal mandibulectomy preserving the inferior border of the mandible was performed. The patient has been followed clinically and radiologically without recurrence for 20 years. Case 2
Fig. 5. Pools of mucus within spinous cell layer and cuboidal cells on surface of epithelium. (Hematoxylin-eosin stain; original magnification, X63.)
A 39-year-old man was seen at the outpatient clinic in 1979 with a left mandibular deformity associated with an intraoral swelling that had been increasing slowly. No pain or suppuration were elicited. Clinically, a massfrom the left canine to the right first molar with expansion of the buccal cortical plate was observed. Radiologically, a multilocular radiolucency from the left second premolar to and including the right first molar was seen. Root resorption was observed (Fig. 1). With the pa-
Glandular
Volume 72 Number I
73
Fig. 9. Case 1. Squamous odontogenic tumor-like epithelium without peripheral palisading. (Hematoxylin-eosin stain; original magnification, X25.)
Fig. 8. Case 3. Epithelial spheres are present in epithehum. (Hematoxylin-eosin stain; original magnification, x63.)
Table
odontogenic cyst
I. Reports of 13 casesof GOC
Case No. (reference)
Localization and x-ray jkdings
Treatment
I
I
Follow-up
1 (2)
69/M
Multilocular radiolucency of anterior mandible
Limited local excision
2 (2)
71/F
Limited local excision
3 (1)
21/F
Multilocular radiolucency of anterior mandible Maxilla, radiolucency
Recurrence of 3 yr; reexcision, no evidence of disease at 18 mo NA
Enucleation and endodontic
No recurrence after 3% yr
4 (1)
59/M
5 (1)
44/F
6 (1)
85/F
7 (1)
59/M
8 (1)
44/M
9 (1) 10 (1) 11
19/M 48/M 45/M
12
52/M
13
39/M
surgery Anterior mandible, unilocular radiolucency Anterior mandible, unilocular radiolucency Mandible, multilocular radiolucency Anterior mandible, unilocular radiolucency Maxilla, unilocular radiolucency Mandible, radiolucency Mandible, radiolucency Anterior mandible, unilocular radiolucency with sclerotic borders Maxilla, well-defined radiolucency Anterior mandible, multilocular radiolucency
Enucleation and endodontic surgery Enucleation NA Curettage Enucleation 9 yr after diagnosis Enucleation Enucleation Marginal mandibulectomy preserving inferior border Maxillectomy Enucleation, endodontics surgery
Recurrence at 3 yr 8 mo; enucleation Recurrence at 3 yr 3 mo; curettage NA No recurrence for 6 mo; lost to follow-up No recurrence in 2 yr No recurrence No recurrence No evidence of disease for 20 yr No evidence of disease for 14 yr No evidence of disease for 10 yr
NA. Not available.
tient under general anesthesia,the cyst was enucleated and root canal procedureswere performed from the right second premolar to the left second premolar (Fig. 2). The patient has beenfollowed without evidenceof disease for 10 years.
Case 3
A 52-year-old man was seen in 1968 for investigation of a gingival tumor. He had gingival swelling and facial asymmetry associatedwith a dull ache over the left anterior maxilla for 6 years. Radiologically, a unilocular radiolu-
74
Patron, Colmenero, and Larrauri
cency was identified, spanning the left lateral incisor to the left first molar without root resorption (Fig. 3). With the presumptive diagnosis of ameloblastoma, a partial maxillectomy was performed. The patient was followed until 1982 without any sign of recurrence. HISTOLOGIC
FINDINGS
The three lesions had the histomorphologic characteristics of the GOC as described by Gardner et al. 1 All lesions had a multilocular gross appearance and showed polycystic features microscopically. Histopathologic examination of the lesions revealed a stratified squamousepithelial lining of varying thickness, with a flat interface with the subjacent stroma (Fig. 4). The connective tissue wall in all caseswas relatively free of inflammatory cells. The superficial layer of the epithelium showed eosinophilic cuboidal and ciliated cells. Pools of periodic acid-schiffpositive, diastase-resistant material were present within the epithelium. Sometimes these mucus pools were lined by eosinophilic cuboidal cells, similar to those found in the lining epithelium (Fig. 5). Cases2 and 3 showed abundant mucous cells on the lining surface and sometimes formed mucous glands (Fig. 6). Occasionally the basal cells were vacuolated (Fig. 7). In case 3 the epithelial cells were arranged into spherical structures (Fig. 8). Case 1 presented islands of squamous epithelium that lacked peripheral palisading and that were in continuity with the cystic epithelial lining (Fig. 9). These squamous epithelial islands were completely confined to the cyst wall. None of our casesshowed calcifications or areas of hyalinization. DISCUSSION
The GOCs presented herein fulfill the diagnostic criteria proposedby Gardner et al.’ From the limited number of casesof GOC that have appeared in the literature (Table I), it seemsthat these lesions have a predilection for men (9/ 13) and for the mandible (lo/ 13). The age range is 19 to 85 years, with a mean age of 50 years. Radiographically the lesions are well defined, with a unilocular or multilocular pattern but without specific features that permit separation from other entities.
ORAL SURGORAL MED ORAL PATHOL July 1991
Of the 13 casesreviewed, 10 were adequately followed up and 3 recurred. The biologic nature of GOC seemsto be intermediate between odontogenic keratocyst and other cysts in size and multilocularity. Enucleation seemsto be adequate treatment despite the few reported cases. The presence of epithelial spheres and squamous odontogenic epithelial proliferations, which have been described in other odontogenic cysts, lends support to the speculation that GOC is odontogenic in nature. We agree with Gardner et al.,’ who suggest that the GOC is a histologic variant of the botryoid odontogenit cyst. The following features are in keeping with a botryoid odontogenic cyst: the occurrence during the fifth to the seventh decadesof life, the intrabody location in the mandibular premolar-canine region, the multilocular radiographic appearance,the recurrence of somelesions, and the presencehistologically of epithelial spheres. Recently Heikinheimo et a1.4found mucous cells in the biopsy specimen of a case of botryoid odontogenic cyst that recurred several times. The relationship, if any, of these two cysts should become clearer as more examples of each are studied.
REFERENCES Gardner DG, Kessler HP, Morency R, Schaffner DL. The glandular odontogenic cyst: an apparent entity. J Oral Pathol 1988;17:359-66. PadayacheeA, Van Wyk CW. Two cystic lesionswith features of both the botryoid odontogenic cyst and the central mucoepidermoid tumour: sialoodontogenic cyst. J Oral Pathol 1987;16:499-504. Wright JM. Squamous odontogenic tumor-like proliferations in odontogenic cysts. ORAL SURGORAL MED ORAL PATHOL 1919;47:354-8.
Heikinheimo K, Happonen RP, Forssell A, Kuusilehto A, Virtamen P. A botryoid odontogenic cyst with multiple recurrences. lnt J Oral Maxillofac Surg 1989;18:10-3.
Reprint requests:
Mercedes Patron, MD Departamento de Anatomia Patologica Hospital La Paz PaseoCastellana 26 1 28046 Madrid, Spain