Table 1.
No. | Reference | Gender/on set age (year) a | Site b | Image features c | Displacement of the teeth/root Resorption d | Biochemistry test e | Treatment | Follow-up | GNAS mutation analysis | HRPT2 mutation analysis |
---|---|---|---|---|---|---|---|---|---|---|
1 |
Bradley and Leake [38], 1968 |
F/6 |
Les. 1: rught maxilla Les. 2: right angle of the mandible |
P.R.: multicystic Lesions |
Yes/N.S. |
N.S. |
Right maxilla:enucleation and curettage right mandible: scheduled for removal |
N.S. |
N.S. |
N.S. |
2 |
Takeda and Fujioka [39], 1987 |
M/55 |
Les. 1: left maxilla Les. 2: right maxilla |
P.R: well-circumscribed lesions showed radiolucent areas mixed with radiopaque areas |
N.S. |
N.S. |
N.S. |
Patient refused treatment |
N.S. |
N.S. |
3 |
Hauser et al [40], 1989 |
M/35 |
Les. 1: right maxillary sinus Les. 2: left maxillary sinus |
P.R.: well-circumscribed mixed radiolucent/radiopaque lesions CT: well-circumscribed lesion with calcified masses |
No/N.S. |
N.S. |
Right maxillary sinus: enucleation left maxillary sinus: partial hemimaxillectomy |
N.S. |
N.S. |
N.S. |
4 |
Yih et al [41], 1989 |
F/31 |
Les. 1: left mandible Les. 2: right maxilla Les. 3: left mandible (2 years later) |
P.R.: well circumscribed unilocular radiolucency |
N.S. |
ALP: 218 IU/L↑Ca : normal limits P: normal limits |
Left mandibular body and right maxilla:enucleation |
Recurrence of the left mandible after 2 y later |
N.S. |
N.S. |
5 |
Khanna and Andrade [42], 1992 |
M/33 |
Les. 1: right maxilla Les. 2: left mandible |
P.R.: large lesions contained diffused calcifications |
N.S. |
ALP: normal limits Ca: normal limits |
Both of the two lesions: enucleation |
Lost for follow-up |
N.S. |
N.S. |
6 |
Hwang et al [43], 2001 |
F/25 |
Les. 1: right mandible Les. 2: left maxilla Les. 3: left mandibuar body Les. 4: left maxilla Les. 5: right maxilla |
P.R.: large calcified mass surrounded by a radiolucent halo zone with corticated margin |
Yes/yes |
N.S. |
Right mandible: partial hemimandibulectomy right maxilla:hemimaxillectomy |
Initially refused treatment; 3 y later, surgical remission of the lesions was undertaken |
N.S. |
N.S. |
7 |
Bertolini et al [44], 2002 |
F/37 |
Les. 1: left maxilla. and hard palate Les. 2: right mandible Les. 3: left mandible |
P.R.: large radiolucency lesions with interspersed calcifications CT: revealed fibrous calcified masses that involved the left maxilla and the right and left mandibular body |
No/no |
N.S. |
Right mandible: partial mandibulectomy Left mandible: curettage Left maxilla: intraoral surgical removal |
Mandible: no recurrencey after 2 y Maxilla.: no recurrence after 1 y |
N.S. |
N.S. |
8 |
Barberi et al [45], 2003 |
F/53 |
Les. 1: left infraorbital region Les. 2: right hard palate |
P.R.: showed partial opacification of left maxillary sinus CT: two different multilocular, inhomogeneously hypodense entities walled in an irregularly thick sclerotic border |
No /no |
N.S. |
N.S. |
N.S. |
N.S. |
N.S. |
9 |
Stergiou et al [46], 2007 |
F/36 |
Les. 1: left mandible Les. 2: right mandible Les. 3: left maxilla |
P.R.: well circumscribed unilocular radiolucency containing diffuse calcifications CT: well demarcated lesions, low density and scattered calcifications |
N.S. |
N.S. |
Enucleation and curettage |
No recurrence after 6 months |
N.S. |
N.S. |
10 |
Chindia et al [47], 2008 |
F/27 |
Les. 1: right angle and body of the mandible Les. 2: left maxilla |
P.R.: mandibular lesion was corticated and maxillary lesion was less well defined with almost complete obliteration of the maxillary sinus |
N.S. |
N.S. |
Both of the lesions: enucleation |
Recurrence after 6 months (mandible) |
N.S. |
N.S. |
11 |
Ribeiro et al [48], 2011 |
F/35 |
Les. 1: left mandible Les. 2: right mandible |
P.R.: large radiolucency surrounded by a radiopaque halo in the left and right mandible CT: unilocular and hypodense image |
Yes/yes |
Ca: 9.73 mg/dl P: 4.2 mg/dL PTH: 56.34 pg/mL |
Both lesions: enucleation |
No recurrence after 3 y |
N.S. |
N.S. |
12 |
Agarwal et al [50], 2012 |
F/20 |
Les. 1: left posterior maxilla Les. 2: right posterior mandible |
P.R.: maxillary and mandibular lesion was well-defined with a radiolucent rim and sclerotic border CT: hyperattenuated masses in left maxillary and right mandibular alveolar ridges |
Yes/no |
N.S. |
N.S. |
Take an operation fifteen years ago of maxilla |
N.S. |
N.S. |
13 |
Popli et al [21], 2012 |
F/19 |
Les. 1: left maxilla Les. 2: right mandible |
P.R.: well-defined mixed radiolucent and radiopaque lesions CT: mixed-density, expansile lesions present at the alveolar process of both the maxilla walled by irregularly thick sclerotic border. |
Yes/yes |
No sign of hyperparathyroidism |
Enucleation |
No recurrence after 2 y |
N.S. |
N.S. |
14 |
Akcam et al [49], 2012 |
M/20 |
Les. 1: left maxilla Les. 2: left mandible |
P.R.: well defined,multilocular radiolucent lesion of the left mandible unilocular radiolucent lesion in the left maxilla CT: extensivehypodense lesions with cortical expansion |
Yes/no |
Ca, P, PTH within normal limits |
Enucleation |
No recurrence after 8 months |
N.S. |
N.S. |
15 |
Kiran Desai et al [22], 2013 |
F/18 |
Les. 1: right maxilla Les. 2: right mandible |
CT: large well-defined expansile lesion, heterogeneously hyper dense, multiple internal punctuate calcifications |
Yes/N.S. |
Blood calcium levels within normal limits, no sign of hyperparathyroidism |
Enucleation |
No recurrence after 2 years |
N.S. |
N.S. |
16 |
Ponniah et al [23], 2013 |
F/45 |
mutiple lesions from the left mandible to the right mandible |
P.R.: multilocular radiolucent lesion CT: osteolytic, soft-tissue density lesion with thinning and erosion of the buccal cortex in the anterior region of the mandible |
Yes/yes |
N.S. |
Enucleation |
No recurrence after 5 months |
N.S. |
N.S. |
17 |
Present case 1 |
F/15 |
Les.1: right maxilla Les. 2, 3: bilateral mandible |
P.R.: well demarcated radiolucency in the bilateral mandible |
Yes/yes |
Ca: 2.7 mmol/L P: 0.77 mmol/L↓ ALP: 278 IU/L↑, PTH not find to text |
Enucleation |
Recurrence after 1 year |
No |
No |
18 | Present case 2 | M/6 | Les. 1: right maxilla Les. 2: left maxilla Les. 3: right mandible Les. 4: left mandible | P.R.:central inhomogeneous mineralizationin thin marginal radiolucent area with a well demarcated sclerotic border CT: mixed radiopaque image | Yes/yes | Ca: 2.63 mmol/L P: 1.58 mmol/L ALP: 62 IU/L PTH 9.12 pg/mL | Incisional biopsies the treatment delayed because of the young age | After 1 year, the lesions enlarged obviously | No | No |
aF = female; M = Male.
bLes. = lesion.
cP.R. = panoramic radiograph; CT = Computerized Tomography.
dN.S. = not stated.
eALP = Alkaline phosphatase; Ca = Serum calcium; P = Phosphate.