- Email: [email protected]
Ossifying fibroma involving the maxilla and mandible
Ossifying fibroma involving the maxilla and mandible Report of a case
E. AS’.Bradley, Jr., D.D.X.,* and Donald Leake, D.M.D.,#* Boston, Mass. DEPARTMENT AND
OF PEDODONTICS,
DEPARTMENT
DENTAL
OF PEDIATRIC
THE
CHILDREN’S
DENTISTRY,
HOSPITAL HARVARD
MEDICAL SCHOOL
CENTER, OF
MEDICINE
0
ssifying fibroma is a lesion of the jaws consisting of fibrous connective tissue and varying amounts of bone. The term ossifying fibroma is controversial. The entity is described in the literature under a variety of names, including fibrous dysplasia of the jaws, fibro-osteoma, localized osteitis fibrosa, localized fibrous dystrophy, fibrous osteoma of the jaws, hypertrophic localized osteitis, localized leontiasis ossea, and monostotic fibrous dysplasia. Controversy arises over theories of histogenesis and interpretation of histopathologic characteristics.l, 2 CLINICAL CHARACTERISTICS AND DESCRIPTION The cause is unknown. Theories include a reparative reaction related to trauma, an abnormal repair process, a neoplastic process, and the result of a developmental defect. Ossifying fibroma involves the maxilla more often than the mandible, it occurs in females more often than in males, and it can occur at any age but is commonly seen in children. In the early stages, few symptoms are noted. Pain seldom occurs. Asymmetry of the face may be one of the first signs. If there is no deformity, the lesion usually is discovered during routine radiographic exam*Resident in Pediatric Dentistry, The Children’s Hospital Medical Center; Teaching Fellow in Pediatric Dentistry, Harvard School of Dental Medicine. Present address: 313 Park Ave., Falls Church, Va. 22040. **Associate in Oral Surgery (Oral Pathology Research), The Children’s Hospital Medical Center.
605
606
Bradley
O.S., O.M. & O.P. Norrmher, 1968
and Leak
ination. There may be diplopia when 6hc eye on the affected side is displaced. The maxilla, the malar bone, the cthmoid and na.sal bones, and the maxillary sinuses may be affected. In the mandible, the lesion may occupy the alveolar process or the entire body of the mandible to the inferior border. In either jaw the lesion grows in an expansile manner and can cause tooth migration. Radiographic
appearance
Radiographic appearance varies with the amount of calcified the lesion. A predominantly fibrous lesion appears as a. well-defined
Figs. 2, 8, rind 8. Patient
Fig. 4. Intraoral
Fig.
Fig. d
Fig. 1
at time of admission.
photographs
showing involvement
of right maxilla.
material in radiolucent
3
Volume 26 Number 5
Ossifying
fibroma
607
area ; as calcification occurs, fine trabeculae or radiopaque areas are seen. Cortical expansion is noted, particularly in larger lesions in the mandible. In the maxilla, the lesion may extend into the sinuses, and its borders may be difficult to discern. Histopathology
Crossly, the lesion is yellowish white in color, encapsulated, and gives a gritty sensation on cutting. The most prominent histologic feature is the presence of fibrous connective tissue in varying degrees of maturity. The tissue may be composed exclusively of young fibroblasts or collagenous tissue. The fibrous tissue tends to be arranged in whorls. The second component of the lesion is calcified material, which varies from isolated foci scattered throughout the fibrous tissue to a dense network of bony trabeculae. The surfaces of the trabeculae show both apposition and resorption. In time, the trabeculae become larger and the fibrous tissue increasingly collagenous, while vascularity, which is initially prominent, diminishes. Treatment
and
prognosis
Because these are slow-growing and often well-defined lesions, the treatment of choice is complete enucleation with curettage. The prognosis is good, since recurrence is rare. These lesions shown no indication of metastasizing or undergoing malignant change. Radiation is of no value and, indeed, is dangerous, particularly in children.
Fig. 5. Preoperative radiograph showing expansion of maxilla and mandible. Note displacement of teeth.
60% Bradley
and Leake
O.S., O.M. & O.P. November, 1968
CASE REPORT A 6-year-old Negro girl who had a painless expanding mass of 4 years’ duration in the maxilla was referred to The Children’s Hospital Medical Center for evaluation. Born of indifferent parents, the child was neglected until an aunt intervened legally and sought medical care. right
Physical
examination
On admission to the hospital, the significant physical findings were marked asymmetry of the right side of the face, with swelling involving the area bounded by the right angle of the mandible, the ear, the inferior border of the right eye, and extending beyond the midline, deviating the nares to the left (Figs. 1, 2, and 3). Intraorally, the right maxillary alveolar ridge was broadened and deviated downward. The swelling involved the hard palate and the soft palate. The teeth in the right maxilla appeared engulfed by the tumor (Fig. 4). On palpation, the tumor was hard, nontender, and fixed. There was limited movement of the mandible. There was no diplopia, in spite of the anterior displacement of the right eye. There was a purulent exudate from the right orbit. The ears were unremarkable. The child weighed 19.5 kilograms. laboratory
findings
The hemoglobin was 12 grams, and the hematocrit count was 8,600, with a normal differential. Bleeding normal. Urinalysis yielded normal values. Radiographic
was 35 per cent. The white blood and coagulation times were also
examination
Radiographs of the skull and facial bones revealed an expansile mass .arising in the right maxilla, with multiloculated and multitrabeculated osseous septa (Fig. 5). Grossly, the mass displaced the teeth posteriorly and cephalad, with loss of the alveolar bone
Fig. 6. Right lateral
cheek flap raised to expose tumor.
Volume 26 Number 5
Ossifying
fibroma
609
support from the displaced teeth. There was obliteration by compression of the maxillary sinuses and elevation of the nasal bone on the right. There was probable obliteration of the airway of the nasal passage, although this was not demonstrable on the film. There was a similar smaller lesion, an estimated 3 by 4 cm., arising from the incisal level of the mandible and producing posterior and superior displacement of the m.andibular incisors. The findings were those of two separate proliferative multicystic lesions (one in the
Fig. 7. Immediate
postoperative
radiograph
showing
space created
Fig. 8
Figs. 8 rind 9. Patient
by removal
of tumor.
F$7. 3
4 months following
removal
of maxillary
tumor.
610
Bradley
and Leake
Fig. 10. Roentgenogram
O.S., O.M. 6; O.P. November, 1968
taken 4 months after
Fig. 11. Lesion showing attached
gingiva
removal
and teeth.
of maxillary
tumor.
Volume 26 Number 5
Ossifying
fibronaa
611
maxilla and one in the mandible) of long duration, with secondary compression and deformity, benign in appearance. A skeletal survey indicated that the bones, joints, and soft tissues were within normal limits. There was no evidence of metastatic tumor. Clinical
course
In the operating room the patient was placed under endotracheal general anesthesia and a biopsy was performed. The tissue section showed fibrillar cells arranged in a whorl pattern, consistent with ossifying fibroma. 3 Because of the extensiveness of the lesion, surgical excision of the maxillary tumor was undertaken. Procedure
With the patient under nitrous oxide, oxygen, and halothane anesthesia with oral intubation, an incision was made through the right philtral crest, around the distorted right maxillary sinus, and the frontal sinuses, and extended posteriorly into the retromolar
E
Fig: 13. Cells as seen at medium-power magnification. Fig. 13. Note appearance of cartilaginous formation
and vascularity.
612
Bradley
Fig.
14
Fig.
15
and Leake
O.S., ox. & O.P. November, 1968
Fig. 14. Acellular concretions are shown at low-power magnification. Fig. 15. Higher-power photomicrograph showing whorl pattern of concretions.
malar area. Next the nose was elevated mesially; an incision was made across the mucosa of both nostrils and a right lateral cheek flap was raised about halfway across the cheek (Fig. 6). Dissection was continued through the infraorbital bone between the soft tissues of the eye and the bone and then back to the right maxillary area along the zygomatic arch. The dissection was then carried along through the right tuberosity area down onto the hard palate. Approximately one half of the mucosa of the hard palate was removed with the tumor. The tumor had dissected its way through lines of least resistance. It involved the sinus system of the face, including the left maxillary sinus, the left ethmoid sinuses, the right maxillary sinus, and the frontal sinuses, and extended posteriorly into the retromolar and pterygopalatine fossa areas. The tumor was removed by means of an osteotome and blunt dissection. A split-thickness graft was cut from the right thigh with a Brown dermatome. The graft lined the cheek flap intraorally, the right maxillary area, the infraorbital area, and the superior part of the mueoperiosteal flap of the hard palate. Four fine mesh packs
Ossifying
Volume 26 Number 5
Fig. 16. Photograph
fibromcl
613
showing bone formation.
were placed in the right maxillary space, the wound was closed, and pressure dressing was applied. Lost blood was replaced with 2,000 C.C. of whole blood. On the fourth postoperative day the patient was returned to the operating room for removal of the four mesh packs in the right maxillary space (Fig. 7). Postoperatively, ocular mobility was surprisingly good and there was no apparent weakness or diplopia. Vision was good. The patient was discharged on the fourteenth postoperative day and was followed at and radiographs taken 4 months l-month intervals on an outpatient basis. Photographs postoperatively indicated good healing (Figs. 8, 9, and 10). Pathology
Gross description. The specimen consisted of two large tumor masses, which were shiny, grayish pink, and moderately firm. The largest mass consisted of tumor with attached gingivae, hard palate, and teeth of the right upper jaw (Fig. 11). The mass measured 7.1 by 7.6 by 4.1 cm. In addition, there were multiple, irregular, smaller fragments of tumor, bone, and teeth. These were gritty when cut and had a white surface. Microscopically, the tissue section showed stellate cells in a compact, somewhat fibrillar stroma. The cells were uniform, with bland nuclei and a moderate amount of cytoplasm (Fig. 12). The stroma was moderately vascular and, in areas, had an almost cartil.aginous appearance (Fig. 13). Acellular concretions were scattered throughout the stroma (Fig. 14). The cells were arranged concentrically about these structures in a whorl pattern (Fig. 15). Bone erosion and focal new bone formation were evident (Fig. 16). The diagnosis was ossifying fibroma of the maxilla.3
DISCUSSION
Historically, pathologists have not agreed on the cause, the clinical course, or the name of this lesion. Yet when three different pathologists independently reviewed the case presented here, their diagnosis was the same-ossifying fibroma.4-” One continues to wonder that a slow-growing lesion could grow to the size of the one presented here without intervention. It is a sad commentary on the
614
Bradley
O.S.,OX. & O.P.
and Leak
November,
1968
social structure of the country and on the indifference of the parents in this particular instance. SUMMARY
A case of ossifying fibroma involving both the maxilla and the mandible has been presented. The maxillary tumor was treat4 by complete enueleation and curettage. The mandibular tumor is scheduled for removal shortly, and reconstruction of the right maxillary arca is planned. The authors wish to acknowledge, with thanks, the encouragement M.D., of the Surgery Service, in presenting this case report.
of Joseph
Murray,
REFERENCES
1. Shapiro, R. D., Francisco, J. V., and Finkelman, Report of a Case, J. Oral Surg. 26: 639-642,1965. 2. Gardner, J. A., and Hanft, R. J.: An Ossifying
A.: Ossifying Fibroma
Fibroma
of the Maxilla;
of the Mandible,
ORAL SURG.,
ORAL MED. & ORALPATH.~~: 128%1294,.1960. 3. Vawter, G. F. (The Children’s Hospital Medical Center, Department of Pathology): Personal Communication. 4. Elzay, Richard P. (Medical College of Virginia, Department of Oral Pathology): Personal Communication. 5. Shklar, Gerald (Tufts University School of Dental Medicine, Department of Oral Pathology) : Person.al Communication. F. Bhaskar, S. N. (Armed Forces Institute of Pathology, Washington, D. C.): Personal Communication.
E. AS’.Bradley, Jr., D.D.X.,* and Donald Leake, D.M.D.,#* Boston, Mass. DEPARTMENT AND
OF PEDODONTICS,
DEPARTMENT
DENTAL
OF PEDIATRIC
THE
CHILDREN’S
DENTISTRY,
HOSPITAL HARVARD
MEDICAL SCHOOL
CENTER, OF
MEDICINE
0
ssifying fibroma is a lesion of the jaws consisting of fibrous connective tissue and varying amounts of bone. The term ossifying fibroma is controversial. The entity is described in the literature under a variety of names, including fibrous dysplasia of the jaws, fibro-osteoma, localized osteitis fibrosa, localized fibrous dystrophy, fibrous osteoma of the jaws, hypertrophic localized osteitis, localized leontiasis ossea, and monostotic fibrous dysplasia. Controversy arises over theories of histogenesis and interpretation of histopathologic characteristics.l, 2 CLINICAL CHARACTERISTICS AND DESCRIPTION The cause is unknown. Theories include a reparative reaction related to trauma, an abnormal repair process, a neoplastic process, and the result of a developmental defect. Ossifying fibroma involves the maxilla more often than the mandible, it occurs in females more often than in males, and it can occur at any age but is commonly seen in children. In the early stages, few symptoms are noted. Pain seldom occurs. Asymmetry of the face may be one of the first signs. If there is no deformity, the lesion usually is discovered during routine radiographic exam*Resident in Pediatric Dentistry, The Children’s Hospital Medical Center; Teaching Fellow in Pediatric Dentistry, Harvard School of Dental Medicine. Present address: 313 Park Ave., Falls Church, Va. 22040. **Associate in Oral Surgery (Oral Pathology Research), The Children’s Hospital Medical Center.
605
606
Bradley
O.S., O.M. & O.P. Norrmher, 1968
and Leak
ination. There may be diplopia when 6hc eye on the affected side is displaced. The maxilla, the malar bone, the cthmoid and na.sal bones, and the maxillary sinuses may be affected. In the mandible, the lesion may occupy the alveolar process or the entire body of the mandible to the inferior border. In either jaw the lesion grows in an expansile manner and can cause tooth migration. Radiographic
appearance
Radiographic appearance varies with the amount of calcified the lesion. A predominantly fibrous lesion appears as a. well-defined
Figs. 2, 8, rind 8. Patient
Fig. 4. Intraoral
Fig.
Fig. d
Fig. 1
at time of admission.
photographs
showing involvement
of right maxilla.
material in radiolucent
3
Volume 26 Number 5
Ossifying
fibroma
607
area ; as calcification occurs, fine trabeculae or radiopaque areas are seen. Cortical expansion is noted, particularly in larger lesions in the mandible. In the maxilla, the lesion may extend into the sinuses, and its borders may be difficult to discern. Histopathology
Crossly, the lesion is yellowish white in color, encapsulated, and gives a gritty sensation on cutting. The most prominent histologic feature is the presence of fibrous connective tissue in varying degrees of maturity. The tissue may be composed exclusively of young fibroblasts or collagenous tissue. The fibrous tissue tends to be arranged in whorls. The second component of the lesion is calcified material, which varies from isolated foci scattered throughout the fibrous tissue to a dense network of bony trabeculae. The surfaces of the trabeculae show both apposition and resorption. In time, the trabeculae become larger and the fibrous tissue increasingly collagenous, while vascularity, which is initially prominent, diminishes. Treatment
and
prognosis
Because these are slow-growing and often well-defined lesions, the treatment of choice is complete enucleation with curettage. The prognosis is good, since recurrence is rare. These lesions shown no indication of metastasizing or undergoing malignant change. Radiation is of no value and, indeed, is dangerous, particularly in children.
Fig. 5. Preoperative radiograph showing expansion of maxilla and mandible. Note displacement of teeth.
60% Bradley
and Leake
O.S., O.M. & O.P. November, 1968
CASE REPORT A 6-year-old Negro girl who had a painless expanding mass of 4 years’ duration in the maxilla was referred to The Children’s Hospital Medical Center for evaluation. Born of indifferent parents, the child was neglected until an aunt intervened legally and sought medical care. right
Physical
examination
On admission to the hospital, the significant physical findings were marked asymmetry of the right side of the face, with swelling involving the area bounded by the right angle of the mandible, the ear, the inferior border of the right eye, and extending beyond the midline, deviating the nares to the left (Figs. 1, 2, and 3). Intraorally, the right maxillary alveolar ridge was broadened and deviated downward. The swelling involved the hard palate and the soft palate. The teeth in the right maxilla appeared engulfed by the tumor (Fig. 4). On palpation, the tumor was hard, nontender, and fixed. There was limited movement of the mandible. There was no diplopia, in spite of the anterior displacement of the right eye. There was a purulent exudate from the right orbit. The ears were unremarkable. The child weighed 19.5 kilograms. laboratory
findings
The hemoglobin was 12 grams, and the hematocrit count was 8,600, with a normal differential. Bleeding normal. Urinalysis yielded normal values. Radiographic
was 35 per cent. The white blood and coagulation times were also
examination
Radiographs of the skull and facial bones revealed an expansile mass .arising in the right maxilla, with multiloculated and multitrabeculated osseous septa (Fig. 5). Grossly, the mass displaced the teeth posteriorly and cephalad, with loss of the alveolar bone
Fig. 6. Right lateral
cheek flap raised to expose tumor.
Volume 26 Number 5
Ossifying
fibroma
609
support from the displaced teeth. There was obliteration by compression of the maxillary sinuses and elevation of the nasal bone on the right. There was probable obliteration of the airway of the nasal passage, although this was not demonstrable on the film. There was a similar smaller lesion, an estimated 3 by 4 cm., arising from the incisal level of the mandible and producing posterior and superior displacement of the m.andibular incisors. The findings were those of two separate proliferative multicystic lesions (one in the
Fig. 7. Immediate
postoperative
radiograph
showing
space created
Fig. 8
Figs. 8 rind 9. Patient
by removal
of tumor.
F$7. 3
4 months following
removal
of maxillary
tumor.
610
Bradley
and Leake
Fig. 10. Roentgenogram
O.S., O.M. 6; O.P. November, 1968
taken 4 months after
Fig. 11. Lesion showing attached
gingiva
removal
and teeth.
of maxillary
tumor.
Volume 26 Number 5
Ossifying
fibronaa
611
maxilla and one in the mandible) of long duration, with secondary compression and deformity, benign in appearance. A skeletal survey indicated that the bones, joints, and soft tissues were within normal limits. There was no evidence of metastatic tumor. Clinical
course
In the operating room the patient was placed under endotracheal general anesthesia and a biopsy was performed. The tissue section showed fibrillar cells arranged in a whorl pattern, consistent with ossifying fibroma. 3 Because of the extensiveness of the lesion, surgical excision of the maxillary tumor was undertaken. Procedure
With the patient under nitrous oxide, oxygen, and halothane anesthesia with oral intubation, an incision was made through the right philtral crest, around the distorted right maxillary sinus, and the frontal sinuses, and extended posteriorly into the retromolar
E
Fig: 13. Cells as seen at medium-power magnification. Fig. 13. Note appearance of cartilaginous formation
and vascularity.
612
Bradley
Fig.
14
Fig.
15
and Leake
O.S., ox. & O.P. November, 1968
Fig. 14. Acellular concretions are shown at low-power magnification. Fig. 15. Higher-power photomicrograph showing whorl pattern of concretions.
malar area. Next the nose was elevated mesially; an incision was made across the mucosa of both nostrils and a right lateral cheek flap was raised about halfway across the cheek (Fig. 6). Dissection was continued through the infraorbital bone between the soft tissues of the eye and the bone and then back to the right maxillary area along the zygomatic arch. The dissection was then carried along through the right tuberosity area down onto the hard palate. Approximately one half of the mucosa of the hard palate was removed with the tumor. The tumor had dissected its way through lines of least resistance. It involved the sinus system of the face, including the left maxillary sinus, the left ethmoid sinuses, the right maxillary sinus, and the frontal sinuses, and extended posteriorly into the retromolar and pterygopalatine fossa areas. The tumor was removed by means of an osteotome and blunt dissection. A split-thickness graft was cut from the right thigh with a Brown dermatome. The graft lined the cheek flap intraorally, the right maxillary area, the infraorbital area, and the superior part of the mueoperiosteal flap of the hard palate. Four fine mesh packs
Ossifying
Volume 26 Number 5
Fig. 16. Photograph
fibromcl
613
showing bone formation.
were placed in the right maxillary space, the wound was closed, and pressure dressing was applied. Lost blood was replaced with 2,000 C.C. of whole blood. On the fourth postoperative day the patient was returned to the operating room for removal of the four mesh packs in the right maxillary space (Fig. 7). Postoperatively, ocular mobility was surprisingly good and there was no apparent weakness or diplopia. Vision was good. The patient was discharged on the fourteenth postoperative day and was followed at and radiographs taken 4 months l-month intervals on an outpatient basis. Photographs postoperatively indicated good healing (Figs. 8, 9, and 10). Pathology
Gross description. The specimen consisted of two large tumor masses, which were shiny, grayish pink, and moderately firm. The largest mass consisted of tumor with attached gingivae, hard palate, and teeth of the right upper jaw (Fig. 11). The mass measured 7.1 by 7.6 by 4.1 cm. In addition, there were multiple, irregular, smaller fragments of tumor, bone, and teeth. These were gritty when cut and had a white surface. Microscopically, the tissue section showed stellate cells in a compact, somewhat fibrillar stroma. The cells were uniform, with bland nuclei and a moderate amount of cytoplasm (Fig. 12). The stroma was moderately vascular and, in areas, had an almost cartil.aginous appearance (Fig. 13). Acellular concretions were scattered throughout the stroma (Fig. 14). The cells were arranged concentrically about these structures in a whorl pattern (Fig. 15). Bone erosion and focal new bone formation were evident (Fig. 16). The diagnosis was ossifying fibroma of the maxilla.3
DISCUSSION
Historically, pathologists have not agreed on the cause, the clinical course, or the name of this lesion. Yet when three different pathologists independently reviewed the case presented here, their diagnosis was the same-ossifying fibroma.4-” One continues to wonder that a slow-growing lesion could grow to the size of the one presented here without intervention. It is a sad commentary on the
614
Bradley
O.S.,OX. & O.P.
and Leak
November,
1968
social structure of the country and on the indifference of the parents in this particular instance. SUMMARY
A case of ossifying fibroma involving both the maxilla and the mandible has been presented. The maxillary tumor was treat4 by complete enueleation and curettage. The mandibular tumor is scheduled for removal shortly, and reconstruction of the right maxillary arca is planned. The authors wish to acknowledge, with thanks, the encouragement M.D., of the Surgery Service, in presenting this case report.
of Joseph
Murray,
REFERENCES
1. Shapiro, R. D., Francisco, J. V., and Finkelman, Report of a Case, J. Oral Surg. 26: 639-642,1965. 2. Gardner, J. A., and Hanft, R. J.: An Ossifying
A.: Ossifying Fibroma
Fibroma
of the Maxilla;
of the Mandible,
ORAL SURG.,
ORAL MED. & ORALPATH.~~: 128%1294,.1960. 3. Vawter, G. F. (The Children’s Hospital Medical Center, Department of Pathology): Personal Communication. 4. Elzay, Richard P. (Medical College of Virginia, Department of Oral Pathology): Personal Communication. 5. Shklar, Gerald (Tufts University School of Dental Medicine, Department of Oral Pathology) : Person.al Communication. F. Bhaskar, S. N. (Armed Forces Institute of Pathology, Washington, D. C.): Personal Communication.