Background
Materials and methods
Case presentation
Case 1
Case 2
Literature review of sporadic multiple ossifying fibroma
No. | Reference | Gender/on set age (year)a | Siteb | Image featuresc | Displacement of the teeth/root Resorptiond | Biochemistry teste | 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 |
Literature review of HPT-JT related OF
Number | Author | Age/gendera | Single/multiple | Location |
---|---|---|---|---|
1 | Kutcher [24] | 22/M | Single | The right posterior mandible |
2 | Iacobone [25] | 26/F | Single | Left mandible ramus |
3 | Yamashita [15] | 18/M | Single | Right maxilla |
4 | Raue [26] | 29/M | Single | Mandible |
5 | Moon [27] | 18/M | Single | Left mandible |
6 | 17/F | Single | The right mandible | |
7 | Teh [28] | 26/M | Single | Left maxilla |
8 | Mallette [29] | 36/N.S.* | Single | Maxilla |
9 | Rekik [30] | 23/F | Single | The right body of the mandible |
10 | Dinnen [31] | 18/M | Single | The right molar region of the mandible |
11 | Cavaco [32] | 18/M | Single | Maxilla |
12 | 31/M | Single | Mandible | |
13 | 23/M | Single | Mandible | |
14 | 21/F | Single | Mandible | |
15 | Wamakulasuriya [36] | 37/M | Single | The left mandible |
16 | 43/M | Single | The left mandible | |
17 | Teh [28] | 54/M | Multiple | Lesion 1: mandible |
Lesion 2: maxilla | ||||
18 | Mallette [29] | 17/N.S.* | multiple | Lesion 1: mandible |
Lesion 2: hard palate | ||||
19 | Schmidt [33] | 37 M | Multiple | Lesion 1: right maxillary canine and premolar areas |
Lesion 2: left maxillary canine and premolar areas | ||||
Lesion 3: left mandible | ||||
Lesion 4: right mandible | ||||
20 | Szabo [34] | 22/M | Multiple | Lesion 1: right maxilla |
Lesion 2: in the right mandible | ||||
21 | Howell [35] | 16/M | Multiple | Lesion 1: right mandible |
Lesion 2: right mandible | ||||
22 | Aldred [12] | 22/M | Multiple | Lesion 1 the right mandible |
Lesion 2: the left mandible | ||||
23 | Fujikawa [37] | 22/F | Multiple | Lesion 1: left maxilla |
Lesion 2:left mandible | ||||
Lesion 3: right mandible | ||||
24 | Cavaco [32] | 13/F | Multiple | Lesion1: maxilla |
Lesion 2: mandible |