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BENIGN FIBRO-OSSEOUS LESIONS OF T H E

MANDIBLE AND MAXILLA

A Rev i ew of 35 Cases

A R T H U RCH MAMAN, B, BCH, DCP (LoND),MC PATH,*

IANSMITH,BDS, LRCP, LRCS (IRELAND),D SR CS ( E N G ) , ~

LAUREN . A C K E R M A N ,D’

Thirty-five benign fibro-osseous lesions of the mandible and maxilla in the

B a n t u were reviewed. Their clinical and radiographic features are very

similar, and it was impossible to make a definitive diagnosis on these grounds

alone. Although there was some overlap in the pathologic picture in 3 cases,

in the remainder it was possible to make a definitive pathologic diagnosis of

fibrous dysplasia, ossifying fibroma, or giant cementoma. There were 6 instances

of chronic osteomyelitis incorrectly diagnosed clinically, and at first patho-

logically, as either fibrous dysplasia or ossifying fibroma.

L I K I C I A N S AND PATHOLOGISTS EXPERIENCEC considerable difficulty in the diagnosis

antl differentiation of the various fibro-osse-

ous lesions involving the mandible and max-

illa. The most frequently encountered en-

tities are fibrous dysplasia, ossifying fibroma,

arid giant cemcntoma. Other lesions some-

what similar include desmoplastic fibroma

antl myxoma. We believe that it is possible

to

accurately separate these lesions and to de-fine their clinical, pathologic, and radiologic

patterns. Focal chronic osteomyelitis of the

mandible may be crroneously diagnosed clin-

ically and radiographically as fibrous dys-

plasia or ossifying fibroma.

We have reviewed 35 cases from the Barag-

waiiath Hospital, a 2,400-bed hospital serving

the needs of about 700,000 Bantu. These cases

occurred over a 13-year period. Clinical his-

tories, radiographs, and the pathologic as-

pects were studied. Individual sections were

conventionally stained with hematoxylin and

I’ntliologist, South African Institute for Medical

Kescarcli. and Bai-:igwanatli Hospital, Johannesburg,Sonth Africa.

t Mnxillo-Facial Surgeon, Barapanath Hospital.T Honorary Visiting Professor of Surgical Pathology,

trniversitv of the \\’itwatersrand; Senior Research As-sociate, South .\frican Institute for Medical Research.

Address for reprints: L. V. Ackerman, RID, Depart-

ment of Surgical Pathology, Barnes Hospital Plaza,

S t . Louis, Missouri 65110.

The authors thank the Director of the South AfricanInstitute for Medical Research, Prof. J. H. S. Gear,for facilities granted; Dr. P. C. Hauptfleisch, Superin-tendent of Baragwanath Hospital, for permission to

publish; and Mr. Max Ulrich for the photographs.Received for publication February 24 , 1970.

eosin. Special stains, including Masson tri-

chrome, silver impregnation for reticulin, and

PAS sections, were also examined under po-

larized light. Table l shows the number of

cases in each category.

During the time period in which these cases

were reviewed, there were 3 cases of myxoma

and 4 cases of ostcosarcoma. Each one of the

group in Table 1 had distinctive characteris-

tics. Some authors13 group these conditionstogether, antl we admit that at times there

is blurring of the diagnosis of giant cenieritoma

and ossifying fibroma. Features of both may

be present in the same case. It i5 possiblc that

they arise from the periodontal membrane

and take on individual characteristics. We be-

lieve that it is important to separate them be-

cause of their different patterns.

FIBROUSYSPLASIA

Of 5 examples of fibrous dysplasia, 4 oc-curred in the maxilla and one in the mandible.

We believe that this disease is a specific path-

ologic entity and that it consists of fiber bone

which persists as a maturation defect. In this

woven immature bone, there is a random

birefringence under polarized light and silver

stain emphasizes the tangled pattern of the

periphery of individual trabeculae (Figs. 1,

2). This pattern persists and no matter how

long the process has been present, there is no

maturation to lamellar bone.9~ 2 The fibrousstromal background may, however, become

more collagenous. T he ground-glass pattern

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304 CANCERugust 1970 Vol. 26

TABLE . Fibro-osseous Lesions of Mandible and Maxilla

No. cases Mandible Maxilla

Fibrous dysplasia

Ossifying fibroma

Giant cementoma

Chronic osteomyelitis

Features of ossifying fibroma and giant

cementoma with one or otherpredominating

fibrous dysplasia

TOTAL

Uncertain ossifying fibroma vs.

radiographically merges gradually into nor-

mal bone, and i t is not possible to completely

enucleate this lesion. With fracture, and in

the cortical areas, lamellar bone may at times

be present.

CASEREPORT

L. L.,a 13-year-old girl, noticed swelling

of the right side of her face for about 4 years.T he swelling was painless, progressed veryslowly, and involved the right maxilla. Radio-graphic examination showed overgrowth ofthe right maxilla and zygoma (Fig. 3). Themaxillary sinus cavity was still present butirregularly reduced i n size. T h e surgical treat-

ment consisted of reducing the mass of theovergrowth to restore the facial contour.Th er e was no evidence of recurrence one yearlater.

CHRONIC STEOMYELITIS

We were surprised to find 6 instances of

chronic osteomyelitis in this group of 35 cases.

These cases were incorrectly diagnosed radio-

graphically as either fibrous dysplasia or os-

sifying fibroma. Five were in the mandible

and one in the maxilla and radiographicallythere were small areas of radiolucency sur-

rounded by areas of increased density. There

were remarkably few clinical signs and symp-

toms. The histopathologic study showed focal

collections of chronic inflammatory cells in-

cluding many plasma cells surrounded by

numerous bony trabeculae. Some of this bone

was fiber bone, but other trabeculae had nu-

merous cement lines with a somewhat pagetoid

appearance (Figs. 4-6). There was also dead

bone in which the trabeculae had emptylacunae. T h e changes in the bone caused the

error in diagnosis and represented a reaction

to the presence of infection. We were not sur-

5

1 1

9

6

1 4

6 5

5 4

5 1

3 1 2

1 1- - -5 19 16

prised by this lack of clinical signs and symp-

toms because it has been previously noted,

particularly in a single vertebra or in a long

bone, that focal chronic osteomyelitis may

produce prominent radiographic changeswith few clinical symptoms.6 I t probably in-

dicates that the organism has a low virulence

with good resistance by the host in the ab-

sence of systemic findings. If attention is paid

only to the bone, it is easy to see how the

diagnosis of fibrous dysplasia or ossifying

fibroma could be made. I n all these cases, as

would be expected, curettage was followed

by complete healing.

CASEREPORT

H., a 25-year-old man, presented with pain-less swelling of the jaw which had been pre-sent for approximately 3 years. There wasmarked swelling of the angle and ascendingramus of the right side of the ,mandible.

Radiologically, there was marked bony ex-pansion of the angle an d ascending ramus,but there was n o sharp line of demarcationfrom the normal bone (Fig. 7). Curettageand a cosmetic trim of the jaw was performed.The lesion healed and no recurrence was re-

ported.

DESMOPLASTICIBROMA

Desmoplastic fibroma is a benign lesion

which occurs most frequently in the long

bones: humerous, tibia, femur, and radius;

bu t i t can also occur in the flat bones and in

the mandible.ll It is a well-delimited lesion

often with accentuated borders, and, micro-

scopically, it has elongated cells of fibroblas-

tic pattern surrounded by collagen fibers dis-

posed in intertwining bundles. It does not

contain osteoid or bone. There is consider-

able variation in the cellularity, and occasion-

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No. 2 FIRRO-OSSEOUSESIONSF MANDIBLEN D M A X I L L A Schrnaman et n l.

FIG. 1 ( t o p ) . Fibrous stromal background in which there are curved trabeculae of woven

FIG. 2 (bottom). Tangled pattern of reticulin fibers, well shown at the periphery of thebone (H and E, x120).

trabeculae of fiber bone (Gordon and Sweet reticulin stain, ~120).

305

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ally theie is local recrudescence in the lesions

showing increased cellulariiy. T he pattern

throughout thc tumor is the same. There are

no cases of desmoplastic fibroma in our series,

but one of us (13.) treated, in 1964, a 46-

year-old Caucasian man with a lesion in the

mandible. It recurred in 1969 and a hemi-

mandiblectomy was performed.

OSSIFYINGIBROMA

Ossifying fibroma of the mandible and max-

illa may produce a fairly large lesion with

distortion and destruction of the bone. It is

often found in young individuals.l.13

In our 11 cases, the ages ranged from 12 to

38 years. Six cases were in the mandible and

5 in the maxilla. It is well delimited and can

be shelled out by the surgeon in contrast to

fibrous dysplasia which cannot be enucleated.

Microscopically, it is made u p of whorlingmasses of well-differentiated connective tis-

sue associated with a variable amount of bone

formation (Figs. 8, 9). This bone is lamellar

bone, and through polarization and with sil-

ver staining, lamellae are laid down in parallel

lines. The trabeculae are often rimmed by

osteoblasts. In the follow-up of our cases, 2recurred.

FIG. 3. Bony overgrowth ofmaxilla and zygoma on the rightside. Although reduced in s ize,the maxillary sinus cavity is still

present.

circumscribed tumor of the body of the man-dible. Although it was rather radiolucent,

there were foci of calcification or ossification

present. The surgical treatment consisted ofenucleation of the lesion. There was no re-currence when last seen.

GIANT EMENTOMA

This lesion is the most controversial of thegroup. It has been called periapical fibrous

tlysplasia, cementoma and cemento-ossifying

fibroma.2,3 , 4, 5 We are not concerned with

this relatively common lesion, which is small

and seldom extends beyond 5 to 6 mm in

diameter, and usually involves the periapical

nevus of anterior mandibular teeth. We are

concerned, however, with the large lesion,

perhaps best designated as giant cementoma.

This forms a well-defined radiolucent mass.

This lesion also may show whorling masses of

fibrous tissue similar to the ossifying fibroma

(Figs. 11 and 12). However, it is distinguished

by the droplet cementum which is PAS posi-

tive, stains red with Masson trichronie, an d po-larizes in the same manner as lamellar bone

except that lines are thinner.* There is no

osteoblastic rimming and no lacunae are pre-

sent. One case recurred after 9 years.

CASEREPORT CASEREPORTS

J., a 33-year-old man, gave a one-year his-tory of swelling of the body of the mandible.At times, it was painful. Radiographic ex-amination (Fig. 10) showed a fairly well-

p., a 24-year-old woman, complained of apainless swelling of the right side of the jawof about 2 years’ duration. On examintion,there was obvious deformity of the body of

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No. 2 FIBRO-OSSEOUSESIONSF MANDIBLEND MAXILLA S chmaman et a l .

FIG. 4 (top). Bony trabeculae in which there is fiber bone as well as lamellar bone. Note

F I G. 5 (bottom). Lymphocytes and plasma cells in fibrous stroma associated with irregular

irregular cement lines and fibrous stroma with scanty inflammatory cells (H and E, x120).

bony trabeculae (H and E, ~ 3 0 0 ) .

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308 CANCER ugust 1970 Vol. 26

FIG. 6. New bone formation with irregular cement lines antl dead bone around a chronicinfiarnmatorv focus.

the mandible but no restriction of mandibu-

lar movement. The roentgenogram showedan expanding lesion of the body of the man-

dible with a micro-multilocular pattern. DISCUSSIONThere w as marked calcification of the lesion

(Fig. 13). At surgery, partia l trimming antlenucleation was performed, i.e., a cosmetic

reduction to restore the facial contours. T he re

has been no recurrence of the lesion in 4 years.

‘ l h descriptions we have given refer to

classic examples of these lesions and these

FK, . 7. Poorly defined expan-sion of jaw which has a finelylotulated appearance. The fociof radiolucency suiroutided byradiodense areas correspond tosmall abscesses (See Fig. 6 ) .

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N o . 2 FIHRO-OSSEOUSESIONSF MA N D I B L END MAXILLA * Schmaman e t a l . 309

cases can normally be distinguished from each types of responses are limited. During infec-

other . One must remember, however, that tions, bone may die and attempts are then

bone is not a static or lifeless structure, and made to form new bone, often onto the old

it responds to various stimuli. However, the trabeculae (appositional bone growth) or

FIGS.8 ( t o p ) an d 9 (bo t tom) . Ossifying fibroma showing lamellar bone formation and osteo-blastic rimming (H an d E, ~ 1 2 0 ) .

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FIG. 10.radiolucentof th e mancalcificationpreent.

Welllesion

idible.or 08

-circumsciof the I

Small folssification

5bedbodyci of

are

new bone may be added to live bone in an

irregular manner. This results in the pagetoid

appearance which should alert one to look

for infection. Superadded infection may of

course occur in an ossifying fibroma or focus

of fibrous dysplasia, but this is uncommon.

We have found one instance in our series. Th e

pathologic features are summarized in Table 2.

T A B L E 1.

Gian t cementoma Chronic osteomyelitisibrous dysplasia Ossifying fibroma

Type of Fiber bone. Lamellar. Rounded or elongated Lamellar bone with

bone No evidence of matura tion May he rimmed by lamina ted masses without irregular cement lines.

May be condensed near

abscess. Dead bone and

appositional hone may

be present.

N o osteoblastic rimming osteoblasts. lacunae.

Variable stromal pattern.ype of Loose-to-compact depending Stromal pattern very Cellular without any mi-

also be linear st reaks of

calcification (which are

probably dystrophic).

There may he area s indis-

tinguishable from ossifying cells.

fibroma.

strom a on age of lesion, i.e.. ther e uniform throughout . totic activi ty. There may Fibrosis may be loose

may be progressive ma-

turation. too cellular.

May he whorled. No t and often contains

lymphocytes and plas-

ma cells. There may

also be masses of plasma

Reticul in Tangled mass of fibers with- Regular larnellae. No value No value

stain out orientation. Borders More parallel lines.

also tangled, irregular. Borders regular.

PAS stain Slightly positive No value Strongly positive No value

Cement Tangled Regular, parallel Concen tric Irregu lar cement lines

lines often resembling Paget' s

disease.

Polarized Random birefringence Parallel lines

light

~ ~~

Thinner lines than ossifying

fibroma in the mature

lesion. Droplet cementum

seldom Dolarizes.

As above

At surge ry Cannot be enucleated. Well delimited and may Can also be enucleated. Cure ttag e and excess

bone shaved down.esection or trimming

necessary for cosmetic

reasons.

be shelled out.

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No. FIBRO-OSSEOUSESIONSF MANDIBLEND MAXILLA - Schinaman et al .

- FIG. 1 ( to f i ) . Cementifying fibroma showing whorled fibrous tissue with rounded and lineardeposits of basophilic material (“cementum”), H an d E, ~120 .

FIG.12 (bottom). Giant cementoma. This PAS stain accentuates the laminated appearance andabsence of lacunae (PAS, ~ 1 2 0 ) .

311

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312 CANCER ugust 1970 Vol. 26

FIG. 13. Multilocular pattern of the jaw tumor in

giant cementoma. This picture could easily be con-fused with chronic osteomyelitis.

Two situations may give rise to confusion.

First, the presence of cortical lamellar bone at

the periphery of the lesion may cause the le-

sion to be diagnosed as ossifying fibroma.

Furthermore, if there is any repair of bone

it has to pass through a stage of fiber bone

and, therefore, fiber bone may be present in

an ossifying fibroma. Th e stromal pattern ap-

pears to be helpful in distinguishing these

various benign lesions. There may be difficulty

in distinguishing between ossifying fibroma

and giant cementoma.

In 3 cases, the microscopic features have

overlapped and shown foci of giant cemen-

toma on one portion of a section and ossifying

fibroma on another. In some cases, one cannot

tell the difference and the only way to be ab-

solutely certain is if one finds the mass right

u p against a tooth in an early stage which

would then indicate that i t is a giant cemen-

toma.

Smith and Zaveletalz felt that ossifying fib-

roma and fibrous dysplasia were similar ex-

pressions of the same pathologic process.

Hamner and his associates8 felt that all these

lesions were of periodontal origin. I n the

giant cementoma, there small irregularly

rounded basophilic masses are suggestive of

cementum, but at times suggest tortured ir-

regular trabeculae. PeplerlO has shown that

in both fibrous dysplasia and ossifying fibroma

the stromal spindle cells are rich in alkaline

phosphatase suggesting that these cells are

functional osteoblasts and not fibroblasts as

generally believed. T he gross appearance ofthe lesions is also not helpful, since they all

feel fibrous with gritty areas or bony areas.

The clinical features of these cases do not

contribute much to the identification of the

lesion. These will be discussed in a later pub-

lication. They all present as swellings which

are usually, but not always, painless. Even

chronic osteomyelitis may be painless6 A help-

ful radioloyic feature pointing to fibrous dys-

plasia of the maxilla is the persistence of a

small maxillary antrum cavity. Chronic oste-

omyelitis may be loculated. Desmoplastic fib-

roma is loculated. Desmoplastic fibroma is

radiolucent while ossifying fibroma and giant

cementoma are also radiolucent with varying

degrees of focal radiosensitivity.

REFERENCES

1. Amies, A,, and Fleming, W. E.: Central ossifyingfibroma of the jaws. Oral Surg. 15:1409-1414, 1962.

2. Bcrnier, J . L ., and Thompson, H. C.: The histo-genesis of the cementoma. A m e r . J. O r t h o d o n t . 32:543-555, 1Y46.

3. Bra.llev, J . L.: hfultiple cementoma. J. Oral Szirg.

4 . Chaudhry, A . P., Spink, J. H., and Gorlin, R. J.:Periapical fibrous dysplasia (cementoma). J. Ora l Surg.

5. Fontaine, J.: Periapical fibro-ostelma or cemen-toma. J. Cnnnd. Dent . Ass . 21:lO-20, 1955.

6. Garcia, A., Jr., and Grantham, S. '4.:Haemato-genous pyogenic vertehral osteomyelitis. J. Bone Jo in tSurg. 42-A:429436, 1960.

7. Gorlin, R. J., Chaudhry, A . P., and Pindhorg,

J. J.: Odontogenic tumors. Cancer 14:73-101, 1961.

2:278-282, 1944.

lG:483-488, 1958.

8. Hamner, J. E., Scofield, H. H., and Cornyn, J.:

Benign fibro-osseous jaw lesions of periodontal mem-

brane origin. Cancer 22:861-878, 1968.

9. Harris, W. H., Dudley, H. R., and Barry, R. J.:T he natural history of fibrous dysplasia. J. B o n e J o t n t

10. Pepler, W . J.: Ossifying fibromas and their rela-tion to fihrons dssplasia, and other tumours. J . P a t h .

1 1 . Rabhan, W . N., and Rosai, J.: Dcsmoplasticfibroma. 1. no ne J o i n t Surg. 50-A:487-502, 1968.

12. Reed, R . J.: Fibrous dysplasia of bone, a reviewof 25 caqes. Ar c h . P a t h . 75:480-495, 1963.

13. %ith, A. C. , and Zavelata A , : Osteoma, ossify-ing fibroma, and fibrous dysplasia of facial and cranial

bones. A h l A Arch Palh . 54:507-537, 1952.

CUT^ 44-A:207-233, 1962.

Ba c t . 79:408-412, 1966.

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