Introduction and Review of the Literature
Materials and Methods
Results
Case no | Year of Dx | Age | Sex | Site | Central/peripheral |
---|---|---|---|---|---|
1 | 1975 | 32 | Male | Not known | Central |
2 | 1978 | 38 | Female | Not known | Central |
3 | 1980 | 50 | Female | Not known | Peripheral |
4 | 1982 | 38 | Male | Mid Mandible | Central |
5 | 1988 | 25 | Male | Mid to post mandible | Peripheral |
6 | 1992 | 23 | Male | Ant to mid maxilla | Peripheral |
7 | 1993 | 39 | Female | Mid to post mandible | Central |
8 | 1993 | 31 | Female | Ant to mid mandible | Central |
9 | 1997 | 44 | Male | Mid to post mandible | Central |
10 | 1998 | 52 | Male | Mid mandible | Central |
11 | 1999 | 49 | Female | Mid maxilla | Central |
12 | 2003 | 32 | Female | Ant mandible | Peripheral |
13 | 2004 | 69 | Female | Mid maxilla | Central |
14 | 2004 | 25 | Male | Maxillary antrum | Central |
15 | 2007 | 48 | Female | Post mandible | Central |
16 | 2008 | 53 | Male | Maxillary antrum | Central |
17 | 2009 | 30 | Male | Post maxilla | Central |
18 | 2010 | 47 | Male | Mid to post mandible | Peripheral |
19 | 2010 | 27 | Female | Ant to mid mandible | Peripheral |
20 | 2011 | 46 | Male | Mid to post maxilla | Central |
21 | 2011 | 49 | Male | Mid to post mandible | Central |
22 | 2012 | 74 | Female | Ramus of mandible | Central |
23 | 2013 | 52 | Male | Mid Mandible | Central |
24 | 2015 | 32 | Female | Ant maxilla | Peripheral |
25 | 2015 | 55 | Female | Maxillary antrum | Central |
26 | 2016 | 34 | Female | Maxillary antrum | Peripheral |
Case no | Epithelium description | Distinct cellular outline | Prominent intercellular bridges | Eosinophilic cytoplasm | Nuclear/cellular pleomorphism | Mitotic figures | Calcifications/ Liesegang rings | Amyloid | Clear cells | Original diagnosis | Review consensus diagnosis | IHC |
---|---|---|---|---|---|---|---|---|---|---|---|---|
1 | Nests | Y | Y | Y | Y | N | Y | Y | N | Typical CEOT | CEOT | |
2 | Nests | Y | N | Y | Y | N | N | Y | N | Typical CEOT | CEOT | ki67 6% |
3 | Small nests and thin strands | Y | N | Y | Y | N | N | Y | Y (focal) | Unusual, maybe CEOT or OD hamartoma | CEOT vs OdF | |
4 | Nests | Y | Y | Y | Y | N | Y | Y | N | Typical CEOT | CEOT | |
5 | Small nests and thin strands | N | N | Y | Y | N | Y (focal) | N | Y (focal) | Unusual, CEOT (preferred) vs OdF | CEOT vs CCOC | ki67 5% |
6 | Small nests and thin strands | Y | N | Y | Y | N | N | Y | Y (focal) | CEOT | CEOT vs OdF | |
7 | Sheets and thin strands | Y | N | Y | Y | N | Y* | N | Y | CEOT, clear cell variant | No consensus | ki67 < 1% |
8 | Sheets and small nests | Y | Y | Y | Y | N | Y | Y | N | Typical CEOT | CEOT | ki67 2% |
9 | Sheets and thin strands | N | N | Y | Y | N | N | Y | N | Unusual OT, CEOT vs OF | No consensus | |
10 | Sheets and thin strands | Y | N | Y | Y | N | Y | Y | Y (focal) | CEOT | CEOT, clear cell variant | ki67 < 1% |
11 | Small nests and thin strands | Y | N | Y | Y | N | N | Y | N | CEOT | CEOT vs OdF/SOC | ki67 2% |
12 | Small nests and thin strands | Y | Y | N | Y | N | Y | Y | Y | CEOT | CEOT | |
13 | Small nests | Y | N | Y | Y | N | Y (min) | Y | Y | CEOT | CEOT | ki67 < 1% |
14 | Small nests | Y | Y | Y | Y | N | Y | Y | Y (focal) | CEOT, maybe arising from dentigerous cyst | CEOT | ki67 1% |
15 | Small nests | Y | Y | Y | Y | N | Y | Y | N | Typical CEOT | CEOT | |
16 | Sheets and thin strands | Y | N | Y | Y | N | N | N | Y | Unusual, maybe CEOT variant | Ameloblastoma with clear cells | |
17 | Small nests and thin strands | Y | Few | ? | Y | N | N | N | N | Unusual, perhaps non-calcifying CEOT | No consensus | |
18 | Small nests and thin strands | Y | In some areas | Y | Y | N | Y | Equiv | Y (most) | Clear cell CEOT | CEOT, clear cell variant | |
19 | Small nests and thin strands | Y | Y | Y | Y | N | N | Y | N | CEOT | CEOT | |
20 | Small nests | Y (few) | N | Y | Y | N | Y (few) | Y | Y (focal) | CEOT | CEOT vs OdF | |
21 | Sheets | Y | N (few) | Y | Y | N | Y | Y | Y | Unusual, perhaps CEOT | No consensus | ki67 < 1% |
22 | Sheets | Y | N (few) | Y | Y | N | Y | Y | N | CEOT | CEOT | |
23 | Small nests and thin strands | Y | N | Y | Y | N | N | Y | N | CEOT | CEOT | |
24 | Sheets and thin strands | N | N | Y | Y | N | Y* (few) | Equiv | n | CEOT | CEOT vs OC with dentinoid | |
25 | Small nests and thin strands | Y | N | Y | Y | N | Y | Y | N | CEOT | CEOT | |
26 | Small nests and thin strands | Y | N | Y | Y | N | Y* | Y | Y (most) | OT, perhaps CEOT | CEOT, Clear Cell variant vs OC with dentinoid | Ki67 < 1% |
Discussion
Authors | Age/sex | Location | Radiographic features | Histopathological findings | C/P |
---|---|---|---|---|---|
CEOT cystic variant | |||||
Gopalakrishnan et al. [26] | 15M | Left posterior maxilla | Unilocular radiolucency with radiopacities | Cyst lining varying from NKSSE to thickened epithelium with characteristics of CEOT | C |
Channappa et al. [48] | 30M | Left posterior maxilla | Unilocular radiolucency with calcifications in association with impacted tooth #13 | Cyst lined by odontogenic epithelium, majority with uniform thickness, with classic features of CEOT | C |
Urias Barreras et al. [49] | 31M | Left Posterior mandible | Unilocular radiopaque/lucent area | Lining of odontogenic epithelium with necrosis, featuring clear cells (PASD positive and osteodentin | C |
Dantas et al. [50] | 22M | Right posterior mandible | Unilocular, mixed radiodensity lesion, root resorption | Microcystic lined by typical CEOT with abundant clear cells | C |
Sánchez-Romero et al. [25] | 42F | Right posterior mandible | Well-defined mixed radiodense lesion in relation to an un-erupted third molar | Microcystic compartments of varying size and occasional clear cells with classic features of CEOT | C |
CEOT clear cell variant | |||||
Abrams and Howell [31] | 50M | Posterior mandible | Unilocular mixed radiodense/radiolucent | Prominent clear cells with classic features of CEOT | C |
Anderson et al. [37] | 68F | Left mandible molar area | Unilocular radiolucent/radiopacity | Prominent clear cells with classic features of CEOT | C |
Oikarinen et al. [59] | 36F | Mandible Left molar to right premolar | Multiloculated radiolucent with radiopaque central region | Prominent clear cells with classic features of CEOT. Amyloid diagnosed under electron microscopy | C |
Yamaguchi et al. [60] | 36M | Right mandible from anterior to premolar region | Unilocular radiolucency | Prominent clear cells with classic features of CEOT. PAS positive granules in clear cells | C |
Ai-Ru et al. [27] | 64F | Anterior mandible | Not recorded | Prominent clear cells with classic features of CEOT | C |
Asano et al. [43] | 44F | Right maxilla | Unilocular, radiolucent area with root resorption | Islands that frequently contained clear cells with typical features of CEOT | C |
Schmidt-Westhausen et al. [36] | 38M | Right premolar to left incisor region in mandible | Radiolucency with diffuse radiopacities in part of the lesion | Central necrosis of large epithelial islands and clusters of clear cells | C |
Hicks et al. [61] | 59F | Right posterior mandible | Unilocular mixed radiolucency and radiopacity | Prominent clear cells with classic features of CEOT | C |
Kumamoto et al. [62] | 14F | Right maxillary 3rd molar region | Unilocular radiolucency impacted upper right 3rd molar | Prominent clear cells, few mitotic figures and typical features of CEOT | C |
Anavi et al. [33] | 27M | Left mandibular canine and first premolar | Unilocular well-circumscribed radiolucency | Sheets of clear cells, amyloid and few small oval calcifications | C |
Germanier et al. [63] | 44F | Right angle of the mandible enclosing the 3rd molar | Multiloculated radiolucency with calcifications | Clear cells in some places and with typical CEOT | C |
Mohtasham et al. [64] | 18M | Right anterior maxilla | Radiolucency with calcification | Scattered clear cells with typical features of CEOT | C |
Rangel et al. [32] | 65M | Right mandible between lateral incisor and canine | Unilocular radiolucency with radio-opacities | Significant portion of cells are clear and other areas with typical features of CEOT | C |
Sahni et al. [65] | 52M | Right maxilla | Mixed radiodensity lesion | Areas of clear cells within epithelial islands and with typical features of CEOT | C |
Chen et al. [66] | 59F | Posterior mandible/ ramus | Unilocular radiolucency | Nests of clear cells in a pseudoglandular pattern. Other areas with typical features of CEOT | C |
Turatti et al [67] | 25F | Left mandible | Unilocular radiolucency with root displacement | Sheets and nests of clear cells with areas of calcifications and amyloid | C |
Rydin et al. [68] | 40F | Left mandible | Unilocular radiolucency with scattered calcifications | Central portion of the tumor composed of clear cells and periphery with typical CEOT | C |
Chatterjee et al. [69] | 73F | Left maxillary molar region | A large mixed radiodense/RL area spearing maxillary antrum | Typical CEOT with Clear cells. PAS positive | C |
Sabir et al. [70] | 63F | Angle of the mandible | Radiolucent lesion in ramus distal to 3rd molar | Almost all islands are clear cells amyloid in between | C |
Júnior et al. [71] | 42M | Mandibular symphysial region | Unilocular radiolucency with patchy radio density | Most clusters with clear cells and abundant small calcifications and amyloid | C |
Wertheimer et al. [35] | 20M | Right maxillary gingiva | Premolar region cup-shaped area | Typical areas of CEOT with some areas with clear cells | P |
Ai-Ru et al. [27] | 32F 47F | mandibular gingiva | No signs of bone involvement | Typical areas of CEOT with some areas with clusters of clear cells | P |
Houston and Fowler [72] | 27M | Gingiva of right posterior mandible | Underlying bone was normal | Prominent clear cells with classic features of CEOT | P |
Orsini et al. [73] | 32M | Maxillary gingiva | NA | Typical areas of CEOT with some areas with clusters of clear cells | P |
Mesquita et al. [74] | 48F | Right maxilla, canine region | NA | Polyhedral and clear epithelial cells associated with amyloid-like deposition | P |
Anavi et al. [33] | 27M | Left mandible | Alveolar crest resorption | Sheets of clear cells, focal mild atypia with amyloid in between cells and clusters | P |
de Oliveira et al. [75] | 43F | Lesion 1: Left mandible Lesion 2: Left maxilla | Superficial cupping in canine area | some clusters are composed with clear cells with typical features of CEOT | P |
Habibi et al. [76] | 70F | Left maxilla | Normal underlying alveolar bone | Typical areas of CEOT with some areas with clusters of clear cells | P |
Gadodia et al. [77] | 18M | Left mandible | Alveolar crest resorption | Scattered clear cells with classic features of CEOT | P |
CEOT Non-calcified with Langerhans cells | |||||
Asano et al. [43] | 44F | Right maxilla | Unilocular radiolucency | Less cellular, clear cells within polyhedral cell clusters. Birbeck granules seen. No calcification | C |
Takata et al. [44] | 58M | Left maxillary canine premolar region | Unilocular radiolucency | Scattered small islands of epithelial cells. Within islands many spherical bodies seen. Amyloid present. S-100 positive. Birbeck granules identified | C |
Wang et al. [78] | 52F | Right maxilla, central incisor canine region | Unilocular radiolucency | Small nests of polyhedral cells and amyloid deposition. Clear cells present. CD1a positive cells are frequent. No calcification | C |
Wang et al. [79] | 38M 39F | Right mandible Left maxilla | Unilocular radiolucency with patchy radiopacities | Small nests and cords of epithelial cells. Few clear cells. Amyloid present. CD1a + , Birbeck granules identified. No calcification | C P |
Afroz et al. [40] | 20F | Right maxilla, lateral incisor area | Normal underlying alveolar bone | Scattered small islands of polygonal cells and occasional clear cells. Amyloid present. No calcifications. Clear cells confirmed as Langerhans cells (S100) | P |
Chen et al. [45] | 40F 58M | Maxilla | Unilocular radiolucency with root resorption Multilocular radiolucency with root resorption | Small nests and cords of epithelial islands with some clear cells. Amyloid present, CD1a + , langerin + , No calcification | Both C |
Tseng et al. [80] | 24M | Left maxilla, canine premolar area | Unilocular radiolucency with root resorption in canine and premolar | Strands and island of epithelial cells and some clear cells. Scant amyloid, CD1a + , No calcification | C |
Santosh et al. [81] | 44M | Left anterior maxilla | Large unilocular radiolucency | Bland epithelial islands with admixed amyloid. CD1a + cells. No calcification was present | C |
Combined epithelial odontogenic tumor. CEOT /AOT | |||||
Damm et al. [18] | 18M 15F | Mandible | Unilocular predominantly radiolucent, one case with radiopacities | A cystic tumor lined with areas of typical AOT. And some CEOT-like areas | C |
Bingham et al. [82] | 14F | Right mandible | Unilocular radiolucent lesion related to impacted first premolar tooth | Cystic tumor with multiple intraluminal nodules. Some typical AOT and others are CEOT. Amyloid positive. Calcifications noted | C |
Takeda and Kudo [83] | 17F | Right maxilla between incisors | Unilocular radiolucent lesion with flakes of radio densities | Encapsulated solid tumor with areas of typical AOT and CEOT. Amyloid positive | C |
Siar and Ng [51] | 13–28 2M, 3F | 3 in maxilla, 2 in mandible | Radiolucent lesion | Thick walled cystic tumor lined with areas of typical AOT and variable amounts of CEOT-like areas | All C |
Ledesma et al. [84] | 10–21 10F, 2M | 9 in maxilla (most canine region), 2 mandible | Radiolucent lesion most related to impacted canine tooth. Some have radiopacities | Typical AOT areas with CEOT-like areas of variable sizes | 11 C 1 P |
Miyake et al. [85] | 16F | Left maxilla, canine region | Radiolucent lesion related to impacted canine tooth | Encapsulated solid tumor composed with areas of typical AOT and CEOT. Amyloid positive | C |
Rosa et al. [86] | 17 | Anterior mandible | Unilocular radiolucent lesion with radio-opacity centrally | A cystic tumor with solid mural nodules with typical AOT and CEOT areas. Amyloid positive | C |
Epithelial cells | Amyloid- like material | Calcification | Clear cells | Langerhans cell | Stromal cells | |
---|---|---|---|---|---|---|
Histochemical stains | ||||||
Congo red | ✔ | |||||
Thioflavin T | ✔ | |||||
PAS | ✔a | |||||
Tryptophan | ✔ | |||||
IHC stains | ||||||
Pan-cytokeratin | ✔ | ✔ | ||||
Cytokeratin cocktail | ✔ | |||||
EGFR | ✔ | |||||
p63 | ✔ | ✔ | ||||
CK7 | ✔ | ✔ | ||||
CK14 | ✔ | ✔ | ||||
CK8 | ✔ | ✔ | ||||
CK13 | ✔ | ✔ | ||||
CK19 | ✔ | ✔ | ||||
Vimentin | ✔ | |||||
Ameloblast-associated protein | ✔ | ✔ | ✔ | |||
Amelotin | ✔ | |||||
Ameloblastin | ✔ | |||||
Amelogenin | ✔ | ✔ | ||||
S100 protein | ✔ | |||||
CD1a | ✔ | |||||
Langerin | ✔ | |||||
Enamelin | ✔ | |||||
Syndecan-1 (CD138) | ✔ | ✔ | ✔ | |||
E-Cadherin | ✔ | |||||
Amyloid A | ✔ | ✔ | ✔ |