JOURNAL OF ENDODONTICS
Copyright © 2001 by The American Association of Endodontists
ZEBRA HUNT
Zebra XX, Part 2
Giovanna Iezzi, DDS, Giovanna Orsini, DDS, Giovanna Petrone, DDS, and Adriano Piattelli, MD, DDS
In the last issue of the Journal, the case of a 67-yr-old woman was
presented. She had a dull, persistent pain of 6-months duration. A
clinical intraoral examination showed the presence of a draining
sinus with purulent discharge in the region of the right first man-
dibular molar. The patient had undergone an extraction in this area
4 months before. A panoramic X-ray showed the presence of
several multiple, bilateral lesions of the mandible (Fig. 1). Peria-
pical intraoral radiographies showed the presence of a mixed
radiolucent-radiopaque lesion in the right mandible and a dense
periapical radiopaque lesion located around the roots of the first
left mandibular molar.
The following pathologies were suggested in the differential
diagnosis: cemento-ossifying fibroma, Paget’s disease, chronic
sclerosing osteomyelitis, hypercementosis, fibrous dysplasia, ce-
mento-osseous dysplasia, and florid cemento-osseous dysplasia.
CEMENTO-OSSIFYING FIBROMA
Cemento-ossifying fibroma is characterized by a slowly grow-
ing, well-circumscribed painless swelling. It affects mainly pa-
tients in the 20 to 40 yr range. Radiographically it is possible to
observe a radiolucent lesion with varying degrees of calcification
and well-defined margins. Some specimens appear largely ra-
diopaque with a narrow radiolucent rim. The posterior tooth-
FIG 1. Panoramic radiography. Mixed radiolucent-radiopaque le-
sions are present bilaterally in the mandible.
424
Printed in U.S.A.
VOL. 27, NO. 6, JUNE 2001
bearing segment of the mandible is most frequently affected.
Females are affected twice as often as males. Root resorption and
root divergence may be present in some cases. Teeth are usually
displaced. In most cases no relationship with tooth apices is
present. Differentiation of this lesion from periodontal ligament-
derived fibro-osseous lesions requires a careful consideration of all
available clinical, radiological, and microscopical data. The com-
bination of clinical data—such as age, race, and radiographic
findings of lesions located in two or more jaw quadrants—should
be enough to establish the correct diagnosis. The radiographic
appearance of the lesions in our patient could be confused with that
of cemento-ossifying fibroma; cemento-ossifying fibroma, how-
ever, affects mainly younger patients and is painless.
PAGET’S DISEASE
Paget’s disease may pass through three progressive stages:
1. Initial or osteolytic, producing a generalized radiolucency
2. Intermediate, both osteolytic and osteoblastic, with ra-
diopaque areas within the generalized radiolucency
3. Mature stage that shows a dense cotton-wool pattern.
Paget’s disease tends to involve multiple bones, and a case
limited to the jaws would be extremely rare. If the mandible is
involved in Paget’s disease both the maxilla and the skull would
show radiographic evidence of the disease.
CHRONIC DIFFUSE SCLEROSING OSTEOMYELITIS
In chronic diffuse sclerosing osteomyelitis the lesion is unilat-
eral, painful, and composed of a single area of diffuse sclerosis
containing small defined osteolytic areas. It has no apparent age or
race predilection and is not necessarily confined to the tooth-
bearing areas and may involve the ramus. In our patient the lesions
were multiple and bilateral.
HYPERCEMENTOSIS
It is completely asymptomatic and usually discovered on routine
radiographs. Premolars and first molars are most frequently af-
fected. Often teeth are bilaterally involved and a generalized form
Vol. 27, No. 6, June 2001 Zebra Hunt 425

may occur. Radiographically an isolated nodule or the character-

istic club-shaped root may be present. In our case these club-
shaped roots were not present and, moreover, the lesion was
painful.

FIBROUS DYSPLASIA

Radiographically fibrous dysplasia shows a mottled radiopaque-

radiolucent pattern; the radiopaque areas may present a ground-
glass appearance. Fibrous dysplasia usually occurs as a painless,
slow-growing, solitary lesion, causing an expansion of the jaw with
a nontender facial asymmetry. Fibrous dysplasia rarely occurs in
multiple quadrants, and affects individuals in the first and second
decades. Although fibrous dysplasia is most frequently an isolated FIG 2. Acellular globular deposits and irregular bone trabeculae are
unilateral lesion, it may be bilateral in either or both jaws. Max- immersed in a highly cellular stroma. Hematoxylin-eosin ⫻100.
illary lesions are more common than mandibular lesions, and often
involve adjacent bones such as the zygoma or sphenoid. The
gradual transition from lesional bone to surrounding bone is re-
flected in the radiographs that typically show absence of a sharp
line of demarcation.
Teeth in the involved area are not displaced and root resorption
is seldom, if ever, associated with fibrous dysplasia. The occur-
rence in a younger age group, the most common maxillary loca-
tion, and the fact that fibrous dysplasia is usually painless go
against a diagnosis of fibrous dysplasia in our patient.

FOCAL CEMENTO-OSSEOUS DYSPLASIA

It is characterized by the presence of an asymptomatic, focal,

mixed radiolucent-radiopaque lesion with ill-defined borders in the
tooth-bearing areas. Lesions are sometimes related to teeth. A
greater frequency is reported in women and in the posterior man-
dible. Mostly affected are the fourth and fifth decades. The ma-
jority of cases do not show bone expansion. The surrounding bone
penetrates into the lesions over some distance, whereas in ce-
mento-ossifying fibroma the lesion is easily demarcated from
surrounding bone. The radiographic aspect of the mandibular le-
sions in our patients could point to a diagnosis of focal cemento-
osseous dysplasia; this lesion is, however, asymptomatic.
FLORID CEMENTO-OSSEOUS DYSPLASIA
Florid cemento-osseous dysplasia is a well-characterized entity
within the group of cemento-osseous dysplasia. It affects almost
exclusively middle-aged, female, black, and mongoloid patients,
but also caucasians or asians may be affected. The disease is
limited to the tooth-bearing areas of the jaws, and no other local-
izations are present. Other areas of the jaws, including the rami, are
not involved in florid cemento-osseous dysplasia, and this may
help to differentiate this lesion from Paget’s disease or fibrous
dysplasia. Several parts of the jaws are involved, and sometimes all
four quadrants. In some patients lesions may persist or develop in
areas where teeth have been removed. The etiology of florid
cemento-osseous dysplasia is unknown. Most patients are partially
or totally edentulous, and patients may be completely asymptom-
atic. The most frequent radiographic sign is the presence of densely
sclerotic, irregular, or lobular masses often symmetrically located
in various portions of the jaws. These masses are interspersed with
less-defined areas of a mixed radiolucent-radiopaque pattern. The
lesions may range from several millimeters to several centimeters
FIG 3. Rounded deposits of cemento-osseous material are found in
a cellular stroma. Toluidine blue ⫻160.
in size. The lesions start as well-delineated radiolucencies and tend
to develop areas of patchy sclerosis within them. In some lesions
it is possible to find a well-demarcated cementoblastoma-like
radiopacity that appears encapsulated. In mature lesions the mar-
gins tend to be diffuse and poorly defined, and a thin radiolucent
margin may be present in some portions. The degree of maturation
from woven bone increases over time. The surface over florid or
expanded lesions shows periosteal new bone with a thickened
periosteum and a thickened irregularly remodeled cortex. If in the
anterior mandible there are remaining teeth, circumscribed peria-
pical lesions, typical of periapical cemento-osseous dysplasia, may
be found. Very important and critical to diagnosis is the fact that
the lesions are centered on tooth roots. The roots may become
resorbed and fuse with the dense cemental masses. The gradual
increase in the size of the lesions and their fusion can transform the
whole mandible to a patchy mixed radiolucency reminiscent of
Paget’s disease. Microscopically, florid cemento-osseous dysplasia
shows an admixture of woven bone, trabeculae, and droplets of
cementum-like calcifications in a fibroblastic stroma; the latter
often present prominent resting and reversal lines. The cementum-
like calcifications often fuse to form coalescing masses. Florid
cemento-osseous dysplasia tends to be more heavily mineralized
than focal cemento-osseous dysplasia, with less fibroblastic tissue
and more osseous or cementum-like tissue.
Asymptomatic patients should, most probably, be kept under ob-
servation by regular radiographic follow-up without any surgical
intervention because the radiographic picture is diagnostic. Patients
426 Zebra Hunt Journal of Endodontics

usually become symptomatic when the sclerotic masses become ex-
posed to the oral cavity; biopsies, or tooth extraction in the involved
portions of the jaws should be avoided. The main symptoms are dull
pain and drainage. A bone expansion is present only in the late stages.
When the lesions are exposed to the oral cavity, due to progressive
alveolar atrophy under a denture or after extraction of teeth in the
involved areas, an inflammatory component becomes present, and the
process is basically a chronic sclerosing osteomyelitis involving dys-
plastic bone. A sequestration of cementum-like masses will occur
slowly, and this will be followed by healing.
The radiographic aspect in our patient points strongly to a
diagnosis of florid cemento-osseous dysplasia. Also characteristic
is the presence of a draining sinus when the lesions became
exposed to the oral cavity.
Microscopic examination of the removed specimen showed
irregular bone trabeculae in a highly cellular stroma (Fig. 2) and
cemento-osseous material immersed in a cellular stroma (Fig. 3).
The definitive pathological diagnosis was florid cemento-osseous
dysplasia.
Drs. Iezzi, Orsini, and Petrone are research fellows, Dental School, Uni-
versity of Chieti, Chieti, Italy. Dr. Piattelli is professor of Oral Medicine and
Pathology, Dental School, University of Chieti, Chieti, Italy. Dr. Piattelli is also
an honorary senior lecturer, Eastman Dental Institute for Oral Health Care
Sciences, London, UK. Address requests for reprints to Professor Adriano
Piattelli, Via F. Sciucchi 63, 66100 Chieti, Italy.

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