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Abstract. The management of patients with juvenile ossifying fibroma (JOF) remains
controversial. To explore the correlations between different treatments and the
patient prognosis, 15 cases of JOF of the jaw were reviewed. Five patients were
male and 10 were female. Patient age at the time of disease onset ranged from 7 to
18 years (mean 10.9 years). Nine tumours were located in mandible and six in the
maxilla. These cases typically manifested clinically as painless swelling of the jaw
(9/15, 60%); 40% (6/15) of the cases were associated with pain, diplopia, stuffy
nose, and/or rapid growth. Images of JOF can show a radiolucent, mixed, or ground
glass-like appearance. Pathological examinations revealed 10 cases of juvenile
trabecular ossifying fibroma (JTOF) and five cases of juvenile psammomatoid
ossifying fibroma (JPOF). In terms of the treatment plan, six patients initially
received radical surgery; nine patients underwent conservative treatment, among
whom six (6/9, 66.7%) had one or more recurrence. At the end of the follow-up
period, 12 patients had no evidence of tumour recurrence and three cases were alive
with a tumour. In summary, surgeons should develop the surgical plan according to
the extent of the lesion, relapse status, growth rate, and family choice, and these
patients should be followed up closely.
# 2015 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
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Table 1. Clinical information, treatment, and follow-up information for the 15 cases of juvenile ossifying fibroma (JOF).
Patient
No./sex
Age at
onset
(years)
Age at
operation
(years)
Conservative treatment
1/M
8
8
Size
4.5 4
Symptoms
Right maxilla
Curettage
Left maxilla
(posterior)
Right mandible
(posterior)
Left maxilla
Curettage
14
19
2 1.5
Swelling
3/M
13
13
54
Swelling
4(R)/M
18
20
32
Swelling, pain
5(R)/F
16
64
Swelling
6(R)/F
12
15
54
Swelling
7(R)/M
15
17
98
8(R)/M
10
12
10 7
9(R)/F
19
98
Swelling,
rapid growth
Swelling,
rapid growth
Swelling,
stuffy nose,
diplopia
19
12 8
Swelling
64
Swelling
11/F
11
11
12/F
6 4.5
Swelling
13/F
32
Swelling
14/F
4 3.5
15/F
11
15
Swelling, pain,
diplopia,
rapid growth
Swelling, pain,
rapid growth
64
Surgery
Swelling
2/F
Radical treatment
10/F
13
Location
Enucleation
72; alive
with tumour
20; alive
with tumour
38; NED
Right mandible
(posterior)
Left mandible
(posterior/angle)
Right mandible
(posterior)
Left mandible
Curettage/resection + fibula
124; alive
with tumour
95; NED
146; NED
Curettage/resection + fibula
105; NED
Curettage/resection + fibula
40; NED
Right maxilla
Curettage/resection + fibula
128; NED
Left mandible
(posterior)
Right mandible
(posterior)
Left mandible
(posterior/angle)
Right mandible
(condyle)
Left maxilla
(posterior)
Resection + fibula
87; NED
58; NED
36; NED
15; NED
Resection
18; NED
Resection
13; NED
Right maxilla
Curettage
Follow-up
(months);
outcome
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Han et al.
Table 2. Radiographic features and pathology of 15 cases of juvenile ossifying fibroma (JOF).
Pathology
JTOF
JPOF
Patient No.
4
6
7
11
1
12
8*
2
13
14*
10
3
5
9
15
Radiographic features
Ground glass opaque, well-defined border
Mixed radiopaque and radiolucent, well-defined
Multilocular radiolucency, well-defined border
Mixed radiopaque and radiolucent, well-defined
Mixed radiopaque and radiolucent, well-defined
Unilocular radiolucency, well-defined border
Multilocular radiolucency, well-defined border
Not available
Mixed radiopaque and radiolucent, well-defined
Not available
border
border
border
border
JTOF, juvenile trabecular ossifying fibroma; JPOF, juvenile psammomatoid ossifying fibroma.
*
JTOF with aneurysmal bone cyst.
Results
Clinical data
Fig. 1. Imaging findings in juvenile ossifying fibroma (JOF): case 15. (A) Preoperative axial CT showing a bulging mass in the maxilla, a ground
glass-like appearance, and central cystic changes. (B) Preoperative sagittal CT showing a mass intruding into the maxillary sinus and root
displacement. There is a high-density shadow in the surrounding area.
371
Fig. 2. Histopathological features of juvenile ossifying fibroma (JOF). (A) Juvenile trabecular ossifying fibroma (JTOF) (case 4) has a cell-rich
fibrous tissue background with cell-containing immature trabecular bone. A layer of hypertrophic osteoblasts, which form a grid-like structure,
surrounds the trabecular bone. (B) Juvenile psammomatoid ossifying fibroma (JPOF) (case 15) has a fibrous stroma containing spherical
mineralized bodies. The bodies are like cementum, with basophilic cores and an eosinophilic exterior. (C) Aneurysmal bone cyst formed within
JTOF (case 8). The capsule is filled with red blood cells.
the ground glass opaque cases were associated with central radiolucent regions
(Fig. 1), and one case had multiple radiolucent regions associated with ground
glass plaques. CT imaging and three-dimensional reconstruction of the different
orientations showed cortical bone destruction and absorption in nine cases (9/13,
69.2%), with adjacent tooth or root displacement in seven (7/13, 53.8%).
Histopathological features
Fig. 3. Imaging findings in juvenile ossifying fibroma (JOF): case 9. (A) The lesion recurred
after multiple maxillary curettages. Axial CT showing multiple transparent shadows accompanied by ground glass-like plaques. (B) CT reconstruction revealing lesions involving the nasal
cavity and orbital cavity, as well as cortical discontinuity. (C) Immediate CT three-dimensional
reconstruction after maxillary expansive resection and fibula muscle flap transplantation
showing ideal recovery of the jaw morphology. (D) Axial CT showing a metal plate of the
correct shape without evidence of recurrence at 1 year and 2 months after surgery.
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Han et al.
Fig. 4. Imaging findings in juvenile ossifying fibroma (JOF): case 11. (A) Preoperative
panoramic radiograph showing a bulging body in the right mandible. (B) Four years after
mandibular segmental resection and iliac bone graft, the mandible is in good shape with no
evidence of recurrence.
373
Fig. 5. Imaging findings in juvenile ossifying fibroma (JOF): case 13. (A) Preoperative panoramic radiograph showing a transparent shadow in the
right condylar region, with clear boundaries. (B) Preoperative coronal CT showing shadows of mixed densities in the right condylar region. (C) Six
months after condylar tumour resection and cartilage repair, the shape of a metal plate is partially seen, and there is no evidence of recurrence. (D)
Postoperative CT reconstruction showing good recovery of jaw shape.
ment plans were developed for each patient in the present study, after taking
various factors into account. These factors
included the site of the lesion, involvement of the surrounding vital tissue, patient age, growth rate, and parental choice.
Of the 15 patients, nine initially underwent
conservative treatment and six received
local radical surgery.
In cases of large or rapidly growing
lesions, among others, complete resection should be chosen as the initial surgery. Additionally, due to the anatomical
structure, maxillary lesions often involve
the nose, eyes, skull, and other tissues;
this makes complete resection difficult.
Meanwhile, with relatively less important structures around mandibular
lesions, segmental resection is not always necessary. Take case 3 for example: the lesion had a limited range, there
were clear boundaries between the lesion
and the surrounding normal bone, and
the jawbone cortex had continuous
edges; in this case enucleation of the
tumour achieved a good prognosis
(Fig. 7).
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Han et al.
Fig. 6. Imaging findings in juvenile ossifying fibroma (JOF): case 8. (A) The mandibular lesion relapsed after curettage. Panoramic radiograph
showing a mixed shadow containing incomplete permanent crowns. (B) The lesion expanded 7 months after a second curettage. (C) Sagittal CT
showing transparent shadows with mixed densities. (D) Axial CT showing significant bulging in the lingual mandibular cheek. (E) CT
reconstruction revealing severe bone destruction and cortical discontinuity. (F) Two years and 1 month after mandibular segment resection and
fibula flap transplantation, there was no evidence of recurrence.
375
References
Fig. 7. Panoramic radiographs of juvenile ossifying fibroma (JOF): case 3. (A) Before surgery,
JOF presented as a single transparent shadow in the right mandible. There were spots of highdensity shapes. The boundaries were clear. The mandibular cortex had continuous edges. The
adjacent roots were displaced. (B) Two years after removal of the tumour, bone had formed at
the lesion site. There was no evidence of recurrence.
growth, where there is pain and displacement of teeth, and for lesions with one or
more recurrence. In addition, as the recurrence rate following conservative treatment is high at up to 66.7% (6/9), longterm follow-up and vigilance is indicated.
Funding
None.
Ethical approval
Not required.
376
Han et al.
Address:
Liqun Xu
Department of Oral and MaxillofacialHead
and Neck Oncology
Ninth Peoples Hospital
Shanghai Jiao Tong University School of
Medicine
Shanghai 200011
China
Tel: +86 21 23271699 5161;
Fax: +86 21 63166731
E-mail: maxilla@sina.com