ORAL AND MAXILLOFACIAL RADIOLOGY Editor: Allan G. Farman Desmoplastic fibroma of the jaw: A case report and review of literature Nasser Said-Al-Naief, DDS, MS, a Rui Fernandes, DMD, MD, b Patrick Louis, DMD, MD, c Walter Bell, MD, d and Gene P. Siegal, MD, PhD, e Birmingham, Ala THE UNIVERSITY OF ALABAMA AT BIRMINGHAM Desmoplastic fibroma is a benign intraosseous neoplasm that is recognized as the intraosseous counterpart of soft tissue fibromatosis in both gnathic and extragnathic sites. It has a propensity for locally aggressive behavior and local recurrence. In the present report, we define the clinicopathological and radiographic features of a desmoplastic fibroma of the mandible in an 8-year-old white boy who initially presented with a 2-month history of a rapidly expanding, painless mass along the right inferior border of his mandible. A critical and comprehensive review of the English language literature is also provided. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101:82-94) Desmoplastic fibroma (DF) is a rare, myofibroblastic tumor, comprising far less than 1% of all bone tumors including benign bone neoplasms. 1,2 It represents the osseous manifestation of aggressive fibromatosis and was first reported by Jaffe in 1958 3 who described multiple sites of involvement in the tibia, scapula, and femur. The first report of gnathic involvement with DF is attributed to Griffith and Irby in 1965, 4 and since then numerous similar cases have been described. 4-54 The surgical approach to the tumor has been a source of controversy, and variable treatment modalities, with different out- comes, have been implemented. The following is a report of a case of desmoplastic fibroma of the mandible supplemented by analysis of clinicopathological and radiographic features of previously documented gnathic DF emphasizing the outcome of various treatment modalities as implemented in clinical practice. CASE REPORT Initial presentation The patient was an 8-year-old white boy who presented to the Oral and Maxillofacial Surgery Clinic at our institution with a 2-month history of a rapidly expanding, painless mass along the right inferior border of his mandible. The patient initially saw his pediatrician who referred him to an oral and maxillofacial surgeon. This surgeon initially felt that the child most likely had a reactive process within the bone, and a follow-up appointment was scheduled for repeat examination and radiographic evaluation. The patient’s mother sought a second opinion from another oral and maxillofacial surgeon and the child was subsequently referred to us. The child and his mother denied any history of trauma. The patient’s past medical history was noncontributory. Clinical examination revealed a slight expansion of the right inferior border of the mandible as well as soft tissue fullness, directly overlying the bony lesion. The remainder of the head and neck examination was within normal limits. A panoramic radiograph showed a disruption of the right inferior cortex of the mandible. The lesion extended from the inferior border in the premolar region. It had a smooth border and was slightly radiopaque (Fig. 1). A computerized tomography (CT) scan demonstrated an 8-mm right mandibular body mass destroying the outer cortex (Fig. 2). Under local anesthesia, a biopsy of the lesion was obtained and submitted for histological evaluation. Histopathology Microscopic examination revealed an ill-defined spindle cell proliferation in a collagenous background with varying a Assistant Professor, Department of Pathology, University of Alabama at Birmingham; Associate Scientist, University of Alabama at Birmingham Center for Metabolic Bone Disease Comprehensive Cancer Center. b Chief Resident, School of Dentistry, Department of Oral and Maxillofacial Surgery, University of Alabama at Birmingham. c Associate Professor and Residency Director, Department of Oral and Maxillofacial Surgery, University of Alabama at Birmingham. d Assistant Professor, Department of Pathology, University of Alabama at Birmingham. e Professor and Director, Division of Anatomic Pathology, Depart- ment of Pathology, University of Alabama at Birmingham; Senior Scientist, University of Alabama at Birmingham Center for Meta- bolic Bone Disease. Received for publication Aug 16, 2004; returned for revision Mar 22, 2005; accepted for publication Mar 27, 2005. 1079-2104/$ - see front matter Ó 2006 Mosby, Inc. All rights reserved. doi:10.1016/j.tripleo.2005.03.034 82 Vol. 101 No. 1 January 2006
Transcript
Vol. 101 No. 1 January 2006
ORAL AND MAXILLOFACIAL RADIOLOGY Editor: Allan G. Farman
Desmoplastic fibroma of the jaw: A case report and review of literature
Walter Bell, MD,d and Gene P. Siegal, MD, PhD,e Birmingham, AlaTHE UNIVERSITY OF ALABAMA AT BIRMINGHAM
Desmoplastic fibroma is a benign intraosseous neoplasm that is recognized as the intraosseous counterpart of
soft tissue fibromatosis in both gnathic and extragnathic sites. It has a propensity for locally aggressive behavior and local
recurrence. In the present report, we define the clinicopathological and radiographic features of a desmoplastic fibroma of the
mandible in an 8-year-old white boy who initially presented with a 2-month history of a rapidly expanding, painless
mass along the right inferior border of his mandible. A critical and comprehensive review of the English language literature
is also provided. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101:82-94)
Desmoplastic fibroma (DF) is a rare, myofibroblastictumor, comprising far less than 1% of all bone tumorsincluding benign bone neoplasms.1,2 It represents theosseousmanifestation of aggressivefibromatosis andwasfirst reported by Jaffe in 19583 who described multiplesites of involvement in the tibia, scapula, and femur. Thefirst report of gnathic involvement with DF is attributedto Griffith and Irby in 1965,4 and since then numeroussimilar cases have been described.4-54 The surgicalapproach to the tumor has been a source of controversy,and variable treatment modalities, with different out-comes, have been implemented. The following is a reportof a case of desmoplastic fibroma of the mandiblesupplemented by analysis of clinicopathological and
aAssistant Professor, Department of Pathology, University of
Alabama at Birmingham; Associate Scientist, University of Alabama
at Birmingham Center for Metabolic Bone Disease Comprehensive
Cancer Center.bChief Resident, School of Dentistry, Department of Oral and
Maxillofacial Surgery, University of Alabama at Birmingham.cAssociate Professor and Residency Director, Department of Oral
and Maxillofacial Surgery, University of Alabama at Birmingham.dAssistant Professor, Department of Pathology, University of
Alabama at Birmingham.eProfessor and Director, Division of Anatomic Pathology, Depart-
ment of Pathology, University of Alabama at Birmingham; Senior
Scientist, University of Alabama at Birmingham Center for Meta-
bolic Bone Disease.
Received for publication Aug 16, 2004; returned for revision Mar 22,
2005; accepted for publication Mar 27, 2005.
1079-2104/$ - see front matter
� 2006 Mosby, Inc. All rights reserved.
doi:10.1016/j.tripleo.2005.03.034
82
radiographic features of previously documented gnathicDF emphasizing the outcome of various treatmentmodalities as implemented in clinical practice.
CASE REPORTInitial presentation
The patient was an 8-year-old white boy who presented tothe Oral and Maxillofacial Surgery Clinic at our institutionwith a 2-month history of a rapidly expanding, painless massalong the right inferior border of his mandible. The patientinitially saw his pediatrician who referred him to an oral andmaxillofacial surgeon. This surgeon initially felt that the childmost likely had a reactive process within the bone, and afollow-up appointment was scheduled for repeat examinationand radiographic evaluation. The patient’s mother sought asecond opinion from another oral and maxillofacial surgeonand the child was subsequently referred to us. The child andhis mother denied any history of trauma. The patient’s pastmedical history was noncontributory. Clinical examinationrevealed a slight expansion of the right inferior border of themandible as well as soft tissue fullness, directly overlying thebony lesion. The remainder of the head and neck examinationwas within normal limits. A panoramic radiograph showeda disruption of the right inferior cortex of the mandible. Thelesion extended from the inferior border in the premolarregion. It had a smooth border and was slightly radiopaque(Fig. 1). A computerized tomography (CT) scan demonstratedan 8-mm right mandibular body mass destroying the outercortex (Fig. 2). Under local anesthesia, a biopsy of the lesionwas obtained and submitted for histological evaluation.
HistopathologyMicroscopic examination revealed an ill-defined spindle
cell proliferation in a collagenous background with varying
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Volume 101, Number 1 Said-Al-Naief et al. 83
degrees of myxoid changes. The architectural arrangement ofthe spindle cells variedwith groups of cells arranged in parallelfashion mixed with short interlacing fascicles and intertwiningstrands (Fig. 3). The spindle cells were generally uniform in
Fig. 1. Preoperative panoramic radiograph depicting a radi-olucent process at the inferior border of the right mandible(arrows).
Fig. 2. A preoperative CT scan showing a lesion of the rightinferior border of the mandible. Note that the lesion haseroded the cortex and is expanding into the soft tissue(arrows).
appearance, with rare mitoses (less than 1/10 high-powerfields), none were atypical (Fig. 3, insert). Residual bonyfragments were noted at the periphery of the section.
Immunohistochemical stainingParaffin-embedded tissue sections, fixed in formalin, were
stained with the following antibodies obtained from DakoCorporation (Carpinteria, CA), according to manufacturerrecommendations: (1) polyclonal rabbit anti S-100 protein,used at a dilution of 1:400; (2) monoclonal mouse antihumansmoothmuscle actin (clone 1A4), used at a dilution of 1:50; (3)monoclonal mouse antivimentin antibodies used at a dilutionof 1:50; (4) monoclonal mouse antihuman muscle-specificactin used at a dilution of 1:50; and (5) monoclonal mouseantihuman Ki-67 antigen (clone MIB-1), an excellent markerof proliferative activity in soft tissue sarcomas.1 Immunohis-tochemistry was performed as previously outlined by us.55
Briefly, 5-mm sections were placed on Plus slides and bakedovernight. Before staining, heat-induced epitope retrieval bymicrowaving the specimen under standard conditions, in thepresence of the Dako Target Retrieval System, was performedin all but the vimentin-stained sections. Slides were then
Fig. 3. A medium power photomicrograph showing the tumorarranged in spindle cell pattern forming short fascicles andbundles with mild swirling (hematoxylin and eosin stain,original magnification 3100). Insert: The spindle cell prolif-eration displayed less than 1 mitotic figure/10 high-powerfields (arrow) (hematoxylin and eosin stain, original magni-fication 3400).
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84 Said-Al-Naief et al. January 2006
stained using the Ventania ES Systems (Ventania MedicalSystems, Inc, Tucson, AZ), although the primary antibodieswere applied manually. This system used 3-amino, a-ethyl-carbazole (AEC) as the chromogen for detection of the avidin-biotin-antibody complex (ABC method). Negative controlswere prepared in parallel by the use of an irrelevant primaryantibody or mouse serum as a substitute for the antibody ofinterest.
The tumor cells showed focal reactivity for smooth muscleactin (Fig. 4), and strong and diffuse immunoreactivity forvimentin (Fig. 5). Tumor cells were nonreactive for S-100protein and muscle-specific actin and the MIB-1 (Ki67) indexwas low (\3%). Based on the clinical, radiological, andpathological appearance, a diagnosis of desmoplastic fibromawas rendered.
Subsequent courseThe patient underwent a resection with wide margins of the
right inferior border of his mandible. This was performedthrough a neck incision. The resection was carried down to theinferior alveolar neurovascular bundle. The bundle, as well asthe developing dentition, was preserved. No grafting wasperformed. Microscopic findings for the resection specimenwere in agreement with the findings in the initial biopsy andwere confirmatory of the diagnosis. The patient tolerated theprocedure well and was discharged on postoperative day 2. Hehas been followed for the past 4.5 years without any clinical orradiographic signs of recurrence. The patient has had excellent
Fig. 4. The tumor cells reacted focally with anti-SMA(smooth muscle actin) antibodies. Note the positive internalcontrol (vessel walls) at the edge of the section (white arrows)(original magnification, ABC, 3100). In the low-powerphotomicrograph, the black arrow highlights the immunore-activity to anti-SMA. This is more clearly seen in the higherpower insert. Insert: Higher power photomicrograph demon-strating focal staining with SMA (black arrow) (originalmagnification, ABC,3200). Nonspecific background stainingdeposits can be seen in the upper left corner of thephotomicrograph.
tooth development with remodeling of the inferior border andno neurosensory deficits (Fig. 6).
Review of the literatureClinical features. A detailed review of the English
literature in the past 40 years has generated a total of 74 casesof gnathic desmoplastic fibromas, as presented in Table I.4-54
There was a modest difference in gender distribution (45% inmales vs 54% in females). Twenty-one percent of patientswere white, 9% were Hispanics, 5% were Asian, and 4% wereAfrican American, whereas race was not documented in 39%of the reported cases. The neoplasm occurred in a wide agerange, extending from the first to the sixth decades of life, with84% of patients being younger than 30 years of age atdiagnosis. The majority of cases (84%) involved the mandibleand the balance (16%) occurred in the maxilla. Approximately70% of the mandibular cases and 83% of the maxillary tumorswere located posteriorly.
Signs and symptoms reported also varied. Sixty-five percentof patients presented with asymptomatic swelling on initialpresentation. Pain, in or around area of involvement, wasreported in 15% of patients, limited mouth opening/trismuswith or without malocclusion in 11%, tooth mobility in 7%,proptosis in only 2.6%, the presence of infection (with orwithout adenopathy) in 2.6%, and an elevation of an ear lobe in1.3% of patients. Dysesthesia and bleeding were reportedseparately in 2.6% of patients. In 19.4% of patients, signs andsymptoms were not documented. One patient8 was excludedfrom the data analysis since no information was provided.
Radiographic features. Conventional radiographicfeatures were reported for 91% of the 74 cases. In general,18% of patients had an ill-defined appearancewith an identicalnumber of cases showing well-defined borders. However thisfeaturewas not emphasized in 61%of cases. Thirty-six percentof patients displayed a multilocular appearance while only 6%showed a unilocular pattern. Only 4% of patients displayed amixed radiolucent/radipaque pattern or a radiopaque pattern.The radiographic features were not further defined in 39% ofcases. One patient was excluded (the last case in reference 32).Where information was available, cortical perforation was
Fig. 5. The tumor cells showed strong and diffuse reactivityfor antivimentin (original magnification, ABC, 3200).
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Volume 101, Number 1 Said-Al-Naief et al. 85
Fig. 6. One-year postoperative panoramic radiograph without evidence of recurrence.
identified in 30% of cases, cortical expansion in 10% of cases,cortical perforation accompanied by fracture in another 3%,and cortical erosion without perforation in only 1 patient. Thetumors caused displacement of teeth, with or without resorp-tion, in 18% of cases. It was also interesting to note that theneoplasmwas reported to occur inmultiple other locations andthus have overlapping radiographic features with other lesionsnative to the gnathic bones.
Management. Patient outcomes were documented in93.2% of cases. Five cases were excluded due to incompleteremoval of the tumor initially,4,20,23 patient refusal of treat-ment,22 or if the treatment rendered was unclear.54
Eleven percent of cases were managed by curettage with orwithout enucleation. Follow-up periods ranged from 5 monthsto 5 years. Sixty-two percent of this cohort showed no evi-dence of disease while 31% of patients exhibited recurrence.Information was not available on 1 patient. Fourteen percent ofcases were managed by excision. The follow-up period rangedfrom 1 to 7 years, during which 10.5% of patients recurred and31.5% of cases did not. No information was available on 2remaining cases. A total of 5.7% of patients were treated byenucleation. The follow-up period ranged from3.25 years to 20years. Seventy-five percent of cases from this cohort exhibitedno evidence of recurrent disease, while no information wasavailable on the remaining case.
The majority of patients (51%) were resected, with 74%showing no evidence of disease during a follow-up period thatranged from 3months to 12 years. Only 4.3% of cases recurredwhile no information was available on 6 patients. A minorityof cases (10.1%) were managed with chemotherapy with orwithout other additional treatment. No recurrence wasexhibited in 85.7% during a follow-up period that rangedfrom 9 months to 9 years. During that period 1 patient waspresumed dead of disease.
Almost all cases demonstrated a classical histologicalpattern, identical to that of extragnathic fibromatosis, ie, anonencapsulated tumor composed of uniformly distributed,
bland, and monomorphic cells, supported by prominentcollagenized to hyalinized connective tissue stroma. It isnoteworthy to mention that scarce reports12,20,21,33 havealluded to the presence of focal hyperchromasia and raremitoses (normal) similar to what we observed.
DISCUSSIONThe World Health Organization defines the histolog-
ical criteria for desmoplastic fibroma as that of a benigntumor of low to variable cellularity, whose cells can beovoid or elongated with uniform nuclei that lack atypia,pleomorphism and mitotic activity.56 The tumor cellsare supported by a matrix of collagenized, variablyhyalinized fibrous connective tissue.56,57 The etiologyof DF remains unknown, however trauma3,58 and endo-crine58 and genetic factors59 have all been suggestedas possible etiologic agents. While most investigatorsbelieve that DF represents the osseous counterpart ofsoft tissue fibromatosis,16,19,25-27 others have classifiedit as a variant of nonossifying fibroma of bone,28 andconsidered it to be biologically intermediate betweenbenign fibrous lesions and fibrosarcoma.60
Our review of the literature revealed a wide age-range distribution as previously reported2,61 but only amodest female predilection,31,58 arguing for equal genderdistribution.38,56 Signs and symptoms are generallynonpathognomonic and onset of symptoms is ofteninsidious, with major destructive lesions often discov-ered at time of initial presentation. On the other hand,pain and swelling are frequently mentioned.62-64
Any bone may be involved with DF,56 but themandible is the most common site of involvement,whereas in extragnathic sites the metaphyseal region offemur, tibia, humerus, or radius, as well as the pelvic
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86 Said-Al-Naief et al. January 2006
Table I. Clinopathological features of gnathic desmoplastic fibroma
Reference
Age/
Gender/
race Site
Radiographic
pattern History Initial treatment
Follow-up
period Comment
Griffith and
Irby 196548 y/$/
white
L mandibular
body and
ramus
MLRL, displacement
of unerupted teeth,
cortical perforation
and pathological
fracture
8-year history of
inability to open
mouth completely,
swelling of L
mandible
Curettage 6 m Tumor was
incompletely
removed.
Marlette and
Gerhard
19685
21 y/#/
NA
L mandibular
angle
MLRL, cortical
expansion
3-week history of
asymptomatic
hard swelling
of L mandible
Curettage 5 y NED
Rabhan and
Rosai,
19686
13 y/#/
NA
Mandible NA 2- to 3-week history of
asymptomatic mass in
mandible
Excision 2 y Suspicious for
recurrence?
Hinds et al.
1969715 y/#/
LA
L mandibular
ramus, Infilt
ration to
surrounding
structures
MLRL, cortical
perforation
1-year history of painful
swelling in L mandible,
decrease in opening and
slight deviation of
mandible to the L on
opening, history of
extraction of mandibular L
3rd molar 3 months prior
Excision, then
radical resection
9 m NED
Schmaman
et al. 1970846 y/#/
white
Mandible NA NA Excision 5 y Recurrence treated with
hemi-mandibulectomy
Gosserez
et al. 197091 y/$/
NA
L mandibular
body and
angle
Ill-defined RL,
cortical
destruction
4-month history of
asymptomatic hard
swelling in L
submandibular region
Horizontal
cervicotomy.
1 y and
8 m
Recurrence treated
by surgical
excision, NED
after 3-month
follow-up
Martis and
Karakasis
197210
30 y/$/
NA
L mandibular
body and
ramus
MLRL 4-year history of facial
deformity and
asymptomatic
progressive swelling in
L mandible with
intraoral swelling
Radical resection/
hemi-
mandibulectomy
NA NA
Dehner 197311 13 y/#/
NA
L mandibular
body
RL 3-week history of
asymptomatic firm
swelling in L mandible
Excision 7 y NED
13 y/#/
NA
L mandibular
body and
angle, soft
tissue
extension
RL 1-year history of
asymptomatic firm
swelling in L mandible
Resection 1 y NED
13 y/$/
NA
L maxilla, palate
and maxillary
sinus
Ill-defined RO
occupying
maxillary
sinus, erosion
of inferior
orbital rim
3-month history of
asymptomatic mass
protruding through socket
of previously extracted
maxillary molar
Resection/subtotal
maxillectomy
2 y NED
Bear 197312 26 y/$/
NA
L mandibular
body and
ramus
MLRL extending
from L bicuspid
to 3rd molar
area, cortical
expansion
2-year history of facial
asymmetry and
asymptomatic
progressive swelling of
mandible extending from
mental foramen area to
mandibular angle, tooth
mobility in the area,
obliteration of L buccal
vestibule intraorally
Curettage NA NA
Hoving a and
Ingenhoes
197413
15 y/$/
NA
L anterior
mandibular
body
Well-defined MLRL
occupying
interproximal area
between R mandibular
lateral incisor and
canine, teeth separated
apart
Few year history of
asymptomatic cherry-size
hard swelling in
R mandibular alveolar
ridge, history of extraction
of a supernumerary tooth
in the area
Resection 3 m NED
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Volume 101, Number 1 Said-Al-Naief et al. 87
Table I. Continued
Reference
Age/
Gender/
race Site Radiographic pattern History Initial treatment
Follow-up
period Comment
Ferguson
1974145 y/$/
NA
R mandibular
angle
Ill-defined MLRL at
mandibular angle,
cortical perforation,
reactive bone
formation
Asymptomatic swelling
in R mandibular
angle
Excision 6 y NED
38 y/#/
NA
R mandibular
ramus
ULRL, cortical
expansion
History of pain in R
mandibular quadrant,
caused by broken, carious,
wisdom tooth, tumor was
incidentally discovered
Enucleation 8 y NED
Badger et al.
19741513 y/#/
white
L mandibular
ramus and
angle
Irregular MLRL,
cortical erosion,
reactive
subperiosteal bone
deposition
4- to 6-year history
of asymptomatic
slowly progressive
swelling of L
mandible, history
of trauma to
area 6 to 8 years
prior
Excision 1 y and
8 m
NED
Cunningham
et al.
197516
4 y/#/
white
R mandibular
body/
submandibular
area
MLRL extended
from 2nd
deciduous molar
up to the
condyle and
coronoid
processes,
cortex thinned
but intact
Several month history of
occasional tooth ache,
3-month history of
hard swelling of
R mandible, intraoral
swelling
Resection/R hemi-
mandibulectomy
1 y1 NED
Sood and
Chatterjee
197517
21 y/#/
In
R posterior
maxilla/
maxillary
sinus
RO of R maxillary
sinus, thickening
of R orbital floor,
thickening of roof
and lateral wall of
R maxillary sinus,
enlargement of
zygoma
4-year history of gradually
progressive swelling in
R maxilla and inferior
orbital margin started
asymptomatic but
developed vague pain
6 months after
Aggressive curett-age/
peripheral
ostectomy
2 y NED
Calatrava and
Donado
197618
2 y/$/
SP
L mandibular
ramus and
angle
Ill-defined RL 2-month history of diffuse
symptomatic swelling
at L pre-auricular
region and
mandibular angle,
intraoral swelling
Chemotherapy, then
hemi-mandibulectomy
2 y? NED. A diagnosis of
low-grade fibrosarcoma
was rendered at first
then a definitive
diagnosis of
desmoplastic fibroma
was established
Fisker and
Philipsen
197619
27 y/$/
NA
L mandibular
body
MLRL, teeth
displaced
2-month history of
facial asymmetry and
swelling at L mandible,
history of trauma to area
5 years previously, teeth
mobility, intraoral
expansion
Curettage 2 y NED
Summers and
Matz
197620
21 y/$/
white
R posterior
maxilla/
maxillary
sinus, soft
tissue
extension
RO of R
maxillary
sinus
Asymptomatic right facial
swelling, expansion of
R maxillary alveolar
process, history of removal
of a similar lesion from
R maxillary sinus 10 years
ago
Enucleation/
curettage
NA NA
59 y/#/
white
L mandibular
ascending
ramus and
condyle, soft
tissue
extension
Well-defined RL
occupying mandibular
ramus and condyle
with extension into
L maxillary alveolar
ridge
Painful intraoral swelling
over L mandibular ramus,
asymptomatic swelling in
the anterior aspect of
alveolar ridge, history of
removal of similar lesion
from the area 10 years ago
Enucleation?
incomplete
removal of
lesion
NA NA, the lesion was
associated with
incidental traumatic
neuroma
Continued
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88 Said-Al-Naief et al. January 2006
Table I. Continued
Reference
Age/
Gender/
race Site Radiographic pattern History Initial treatment
Follow-up
period Comment
Nussbaum
et al.
197621
3 y/$/
white
L mandibular
body from
parasymphysis
to the
ramus
Large RL extending
from parasymphysis
to ramus
3-month history of
asymptomatic progressive
L mandibular swelling,
intraoral buccal and
lingual cortical expansion
Resection/hemi-
mandibulectomy
1 y1 NED
Wagner et al.
19772225 y/#/
LA
L mandibular
body and
ramus
MLRL extending from
premolar area to
ramus, cortical
perforation
Facial asymmetry and
swelling at inferior
border of mandible
Refused treatment N/A N/A
Freedman
et al.
197823
26 y/$/
AA
L mandibular
body at
premolar and
molar area
Well-defined ULRL with
interradicular
extension at premolar
and molar area, root
divergence and
resorption, inferior
displacement of the
inferior alveolar canal,
focal cortical
perforation
4-month history of
asymptomatic L moderate
mandibular swelling with
intraoral swelling in
mandibular buccal
vestibule, teeth mobility
Curettage 6 m Residual tumor present,
patient remained
asymptomatic 4 years
after, no further
treatment was
rendered
Taguchi and
Kaneda
198024
27 y/$/
Or
L mandibular
body, ramus,
and condyle
Ill-defined RL extending
from lower R 1st
molar to the ascending
ramus, cortical
destruction
History of L mandibular
swelling, deviation of
mandible to the R on
opening, TMJ arthralgia
and crepitus with
malocclusion
Hemi-mandibulectomy 1 y and
8 m?
NED
Osguthorpe
et al.
198125
41 y/#/
white
L posterior
maxilla/
maxillary sinus
and pterygoid
plates, focal
soft tissue
extension
Large RL involving L
maxillary sinus with
destruction of sinus
walls erosion
of pterygoid plate
3-month history of
progressive L infraorbital
hypoesthesia, nasal
stiffness with firm
asymptomatic cheek
swelling, proptosis
Maxillectomy/
modified en
block resection
9 m NED
Green and
Gaffney
198126
23 y/$/
white
L mandibular
body and
ramus
Well-defined MLRL,
teeth displacement
6-month history of
asymptomatic cystic
lesion of L mandible with
expansion of L posterior
mandibular alveolar ridge
Resection 1 y and
8 m
NED
Slootweg and
Muller
198327
3 y/$/
white
Posterior
mandibular
inferior
border
Well-defined ULRL
adjacent to an
unerupted tooth.
6-month history of
mandibular swelling
Enucleation 20 y NED
2 y/$/
white
Posterior
mandible
Well-defined ULRL with
suggestion of
pericoronal
location
3-month history of
mandibular swelling
Excision with
curettage
2 y Recurred, treated with
resection then 2nd
recurrence after 5
years treated with
block resection, NED
after 15 years
follow-up
Siemssen and
Anagnostaki
198428
18 y/$/
NA
R mandibular
body and angle
Ill-defined
MLRL
Asymptomatic lesion in
right mandible,
discovered by routine
radiographic
examination
Enucleation, then
wide excision.
1 y and
4 m
NED
Eisen and
Butler
198429
46 y/#/
AA
R posterior
maxilla/
maxillary
sinus
Ill-defined mixed
RL/RO of the R
maxillary sinus
4-year history of
asymptomatic R facial,
lateral nasal, and labial
vestibule swelling
Excision 1 y NED
Addante and
Laskin
198530
6 y/#/
NA
R mandibular
body and
submandibular
area
extending
to midline
Well defined RL, cortical
perforation
1-month history
of asymptomatic
R submandibular
swelling
En bloc resection/
segmental
mandibulectomy
2 y NED
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Volume 101, Number 1 Said-Al-Naief et al. 89
Table I. Continued
Reference
Age/
Gender/
race Site Radiographic pattern History Initial treatment
Follow-up
period Comment
George et al.
19853122 y/#/
white
R maxilla,
beneath the
zygoma
Mixed MLRL/RO lesion
in interradicular area
2nd premolar/3rd
molar, loss of lamina
dura, obliteration of
floor of maxillary
sinus, cortical
perforation
4-month history of
asymptomatic slowly
enlarging swelling in R
maxilla including R
maxillary alveolar ridge
and vestibule, teeth
mobility and displacement
Curettage 1 y NED
Ayala et al.
1986322 y and 3
m/#/
LA
L mandible NA 8-month history of slowly
progressive swelling in
L mandible
Chemotherapy with
partial response,
then excision, then
chemotherapy again
4 y and
3 m
NED
3 y and
7 m/$/
white
L mandible Destructive RL 2-month history of
asymptomatic mass
in L mandible
Partial excision, then
chemotherapy with
good response, then
additional
excision and
then more
chemotherapy
9 y and
5 m
NED
2 y and
9 m/#/
white
R maxilla NA 2-week history of painful
mass in R maxillary molar
area simulating an
odontogenic infection
Total resolution of
tumor with
chemotherapy alone
6 y and
4 m
NED
3 y & 7m/
$/LA
L mandible NA 4-month history of
asymptomatic mass in
L mandible.
Excision, then partial
mandibulectomy
with positive
margins, followed
by total resolution
of
tumor with
chemotherapy
7 y NED
4 y and
9 m/$/
LA
Posterior
Mandible
NA 3-week history of swelling in
mandibular molar
simulating an odontogenic
infection
Chemotherapy with
good response, then
conservative
resection
1 y NED
9 y and
11 m/$/
LA
R posterior
maxilla,
extension into
skull base and
nasopharynx
CT: large mass,
involving R skull
base, with
nasopharyngeal
extension and
sphenoid destruction
4-month history of earache
and difficulty in chewing,
mass in R maxilla
discovered following tooth
extraction
Chemotherapy for 7
months with no
response
(uncontrolled
diseases)
9 m then
lost to
follow-
up
9 m alive with disease
then lost to follow-up,
presumed dead from
disease
Bertoni et al.
1986336 y /#/
white
R mandibular
ramus
MLRL, cortical
perforation
1-month history of
asymptomatic facial
swelling
Aggressive
curettage
9 m Recurrence, treated by
excision/resection,
NED after 3 years.
1 y and
10 m/$/
white
R mandibular
body, condyle,
ramus, and
angle
Well-defined RL, cortical
perforation
1-month history of
asymptomatic facial
swelling
Marginal
resection
2 y and
10 m
NED
30 y/$/
white
R mandibular
body,
premolar/
molar area
Ill-defined RL, focal
cortical perforation
1-year history of
asymptomatic facial
swelling
Aggressive curettage 6 y NED
Hietanen et al.
19863413 y/#/
NA
R mandible,
canine
premolar area
Ill-defined ULRL in
interradicular area,
teeth displacement
Asymptomatic swelling in
alveolar crest discovered
during routine
examination, teeth
displaced
Excision NA NA
Makek and
Lello
198635
2 ½ y/#/
NA
R mandibular
body and
ramus
MLRL 18-month history of
asymptomatic R facial
swelling from distal tooth
germ to the ramus, R
mandibular deviation and
malocclusion
Resection 4 y Recurrence, treated by
resection, 2nd
recurrence after 4
years treated with
resection. NED after
27 years follow-up,
parasthesia present
Continued
OOOOE
90 Said-Al-Naief et al. January 2006
Table I. Continued
Reference
Age/
Gender/
race Site Radiographic pattern History Initial treatment
Follow-up
period Comment
33 y/$/
NA
R mandibular
body, ramus
and angle.
Ill-defined MLRL from R
premolar to sigmoid
notch area, cortical
perforation
2-month history of
asymptomatic swelling in
R mandibular angle
Enucleation and
local excision
3 y NED, mild
hypoesthesia
present.
15 y/#/
NA
L mandibular
ramus and
angle
MLRL extending from
3rd molar to
mandibular ramus and
angle
2-month history of
asymptomatic progressive
swelling of L mandibular
ascending ramus,
intermittent pain, elevation
of L ear lobe, intraoral
expansion
Resection 11 y NED, anesthesia exists.
Schultz et al.
1986367 y/$/
NA
L mandibular
ramus
Well-defined MLRL,
cortical destruction
Several weeks’ history of
swelling in L mandible,
limited mouth opening
Curettage 5 m Recurrence,
treated by
excision
Rubin et al.
1987377 y/$/
AA
L mandibular
body and
angle
Well-defined MLRL 1-month history of moderate
swelling in L mandibular
body and angle, trismus,
intraoral swelling in
mandibular vestibule
Aggressive
curettage
10 m Recurrence, treated by
aggressive curettage,
recurred again after
1 year, treated by
hemimandibulectomy,
NED after 1-year
follow-up
Kwon et al.
1989389 y/#/
NA
R mandibular
body and
ramus
Large RL in the
mandibular body and
ramus in second
premolar and molar
areas, teeth displaced
3-week history of
asymptomatic R facial
swelling
Curettage 9 m Recurrence with
symptoms, treated
with partial
hemimandibulectomy,
NED after
approximately 4-year
follow-up
De Vito et al.
19893916 m/$/
NA
L mandibular
body/inferior
mandibular
border
Destructive RL, cortical
perforation
6-month history of
progressive L mandibular
swelling and facial
asymmetry
Curettage 1 y NED
Valente et al.
19894029 y/$/
white
R mandibular
body
Irregular RL apical to
endodontically treated
lower R second
premolar, apical root
resorption
5-year history of a repeated
abscess like lesion apical
to a tooth simulating an
odontogenic infection
En bloc
resection
2 y NED
Christiansen
1990418 y/$/
white
L mandibular
body, ramus,
and condyle
ULRL distal to
unerupted 2nd molar,
inferior alveolar nerve
involvement, cortical
perforation
Asymptomatic swelling of
left mandible, lower left
lip parasthesia, deviation
of mandible to the L on
opening
Resection 1 y Recurrence treated with
resection, recurred
again after 1 year
treated again with
resection, NED after 3
year follow-up
Boon et al.
19914251 y/#/
Ind.
R mandibular
angle and
ramus
MLRL, cortical
perforation with
pathological fracture
of lower mandibular
border, root resorption
6-month history of painful
progressive swelling in R
mandible, local
lymphadenopathy,
intraoral swelling
Resection 1 y NED
Vally and
Altini
199043
NA/$/
white
Posterior
mandible
RL Slow-growing asymptomatic
hard swelling in posterior
mandible
Resection NA NA
42 y/#/
AA
Posterior
mandible
RL Slow-growing asymptomatic
hard swelling in posterior
mandible
Resection NA NA
10 m/#/
AA
Posterior
mandible
RL Slow-growing asymptomatic
hard swelling in posterior
mandible
Resection NA NA
21 y/$/
AA
Posterior
mandible
RL Slow-growing asymptomatic
hard swelling in posterior
mandible
Resection NA NA
12 y/#/
AA
Posterior
mandible
RL Slow-growing asymptomatic
hard swelling in posterior
mandible
Resection 1 y and
6 m
NED
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Volume 101, Number 1 Said-Al-Naief et al. 91
Table I. Continued
Reference
Age/
Gender/
race Site Radiographic pattern History Initial treatment
Follow-up
period Comment
22 y/$/
AA
Posterior
mandible.
RL Rapidly growing
asymptomatic hard
swelling in posterior
mandible
Resection 6 y and
6 m
NED
27 y/#/
AA
Maxilla RL Slow-growing asymptomatic
hard swelling in maxilla
Resection 4 y NED
32 y/$/
AA
Posterior
mandible
RL Slow-growing asymptomatic
hard swelling in posterior
mandible
Excision 1 y NED
Hashimoto
et al.
199144
15 y/#/
Or
L maxilla and
maxillary sinus
RL, indistinct maxillary
sinus floor and
posterior wall, cortical
perforation, root
displacement and
resorption
1-year history of
asymptomatic swelling, L
maxillary buccal vestibule
and molar region,
infraorbital swelling,
palatal displacement and
extrusion of L. maxillary
molar, history of trauma to
area
Resection 7 y NED
Sleeman
et al.
199345
29 y/#/
NA
R posterior
maxillary
alveolar ridge
distal to
premolars
Ill-defined RL in R
posterior maxilla,
teeth destruction
1-week history of
asymptomatic spontaneous
bleeding from R maxillary
quadrant, inflamed
attached gingiva, sinus
tract was also present
En block resection 18 m NED
Cranin et al.
1994462 ½ y/#/
AA
R mandibular
body from
molar to canine
region
MLRL, cortical
thickening
1-month history of
asymptomatic firm
swelling of R mandible
Resection 12 y NED
9 y/#/
white
L mandibular
body and
ramus
ULRL from molar area
to the ramus, teeth
displacement, stunted
growth of condyle
head
2-year history of delayed
eruption of L mandibular
molar, otherwise negative
findings, the tumor exists
for 5 or more years?
en-block resection 2 y NED
Miyamoto
et al.
199547
29 y/$/
Or
L. mandibular
body, incisors
and premolar
area
Well defined ULRL,
cortical thinning and
expansion
3-month history of facial
asymmetry and
asymptomatic swelling in
L mandibular incisors and
premolar area
Curettage with
peripheral
ostectomy
3 y NED, history of tuberous
sclerosis with mental
retardation,
angiomyolipoma of
the kidney, cerebral
calcification, facial
adenoma sebaceum
Iwai et al.
1996483 y and 9
m/$/
Or
L mandibular
body &
ramus
MLRL extending from
area of 2nd primary
molar to ramus,
cortical destruction,
periosteal reaction
with sun-ray like
pattern
2-week history of
asymptomatic firm
swelling in L mandible
Resection 6 y NED
Hopkins
et al.
199649
13 y/#/
NA
R posterior and
anterior
mandibular
body
Well-defined ULRL,
cortical expansion
3-month history of gradual
swelling of R Mandible
Resection 10 m then
lost for
long-term
follow-up
NED
19 y/$/
NA
R mandibular
body and
parasymphysis
ULRL, slight cortical
expansion
Asymptomatic swelling
discovered on routine
examination
Partial resection 1 y and
7 m
NED
Templeton
et al.
199750
6 y/$/
NA
R mandibular
body and
ramus
Ill-defined MLRL,
cortical perforation,
root resorption
10-month history of rapidly
progressive trismus
unresponsive to
orthodontic management,
mildly tender swelling of
R mandibular ramus and
diffuse firm mass in the R
posterior mandibular
vestibule, slight deviation
of mandible to the R with
opening
Continuity/segmental
resection
2 y and
6 m 1
NED
Continued
OOOOE
92 Said-Al-Naief et al. January 2006
Table I . Continued
Reference
Age/
Gender/
race Site Radiographic pattern History Initial treatment
MLRL, multilocular radiolucency; ULRL, unilocular radiolucency; RL/RO, mixed radiolucent and radioopaque; y, years; m, months; NA, not available;
NED, no evidence of disease.
bones, are well reported as other primary sites.62,65 In areview of 184 cases, DF most commonly involved themandible (22%), followed by the femur (15%), pelvicbones (13%), radius (12%), and tibia (9%).2
In general, the radiographic features of DF arenonspecific. These include a unilocular or multilocular,well-demarcated or irregular radiolucency with variablyexpressed marginal sclerosis.19 Therefore, DF oftenmimics other common as well as unusual pathologiesof the jaws including ameloblastoma, odontogenicmyxoma, aneurysmal bone cyst, chondromyxoidfibroma, central hemangioma, and eosinophilic gran-uloma.49 Other unusual patterns have been also described.These include periapical pathosis in association with anendodontically treated tooth,40 and in an interradicularposition at presentation.31
A sunray radiographic presentation, mimickingosteogenic sarcoma48 may be especially significantbecause, although it has been previously described inbenign intraosseous tumors,59 it may wrongly lead to adiagnosis of malignancy. Further, the rapid growth andbone destruction often seen in association with DF,coupled with a spindle cell histological pattern that may
be similar to that seen in a low-grade central osteosar-coma,59 may further lead to an inaccurate diagnosis.Magnetic resonance imaging (MRI) has been shown tocontribute little to the differential diagnosis; however,due to its clear separation of intraosseous tumors fromnormal bone marrow, MRI is most valuable in surgicalplanning,2 while CT is considered superior to MRI indemonstrating the cortical breakthrough seen in 29% ofpatients.66
Immunohistochemical stains may not be alwayshelpful in distinguishing this neoplasm from otherspindle cell tumors and tumor-like lesions that involvethe oral and maxillofacial bones. The tumor cells maynot be reactive with antibodies directed against smoothmuscle actin and muscle-specific actin,62 but immuno-reactivity with the vascular markers anti CD34 andCD31 has been occasionally reported.67-69 In the presentcase, the neoplasm did not stain with vascular markers(with well-delineated positive internal controls, high-lighting the vascular structures in the section), whilehighlighting the low proliferative activity (MIB-1) ofthe lesion making a malignant lesion such as fibrosar-coma less likely.
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Volume 101, Number 1 Said-Al-Naief et al. 93
Arguably, low-grade fibrosarcoma is the most chal-lenging differential diagnosis and the most importantlesion to differentiate from DF. Unlike fibrosarcomawhere cells typically assume fascicular growth patternand often produce a so called ‘‘herring bone’’ appear-ance, DF favors a single cell orientation andmay be seenarranged in bundles. Additionally, overlapping of thespindle cells, increased mitotic activity, pleomorphism,and paucity of collagenous background are character-istic features of fibrosarcoma, and not DF. Anotherfeature that favors a diagnosis of desmoplastic fibromais the presence of indistinct cell borders and a cytoplasmthat merges with the supporting collagenous back-ground. The cells also often contain multiple, yet smallnucleoli. In certain circumstances, the distinctionbetween the 2 conditions may not be possible and allcases must be followed up carefully.1,2,69
It has been recommended that a generous diagnosticbiopsy should be taken from the center of the lesionrather than the periphery in order to avoid misinter-preting the presence of reactive bone at the periphery asosteoid, which may in turn lead to a misdiagnosis ofbenign fibroosseous lesion or osteosarcoma.1,64
Solitary congenital fibromatosis (infantile myofibro-matosis) of bone is most commonly seen in thecraniofacial bones of patients 2 years old or younger.It may be distinguished from DF by its more nodulargrowth pattern and differing immunophenotype.70
Microscopically, DF arising within bone cannot beseparated from extra-abdominal desmoid tumor of softtissue (fibromatosis) infiltrating bone. In these cases,clinical and radiographic features are critical in theseparation of these 2 entities.
Variable treatment modalities have been used for DFincluding surgery, radiation therapy,4,26 and chemother-apy with or without additional procedures.32 Radiationis not recommended since it has been shown to be onlyrarely successful and, because of its potentially muta-genic effect,63 may lead to postradiation sarcoma. Thesurgical approach to the lesion has been a source ofcontroversy. While some surgeons prefer curettage,others prefer wide local excision or recommend resec-tion with a wide margin. Iwai et al.48 reported thatpatients who were treated with resection or wideexcision showed no recurrence, whereas the recurrencerate in those treated with simple excision or enucleationwas 20% to 40%, as compared to 70% recurrence rate inthose treated with curettage alone. Some authors havealluded to the fact that the high recurrence of DF is notonly a result of inadequate surgical excision but alsois a function of the innate biology. Kwon et al.38 re-ported that tumors with high cellularity have higherrecurrences than those with lower cellularity. Given therecurrence rates of DF, the current accepted minimum
follow-up period is no less than 3 years.59 Our reviewhas confirmed that the method of surgery affects therecurrence rate. Resection and excision are superiorover curettage in minimizing postsurgical recurrence,which was significantly lower in the resected cases,when compared to ones treated by curettage. Interestingly,our review of the literature has also alluded to thepresence of subtle atypia or mitotic figures, which wasalso demonstrated in the present case. In the currentcase, we present the management of a moderatelycellular DF, which was treated with a wide localexcision. The patient has been followed in our clinicfor the past 4.5 years without recurrence.
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