The radiographic characteristics of

histiocytosis X
A study of 29 cases that involve the jaws

M. Dagenais, DMD, Dip Oral Radiology,a A4.J. Pharoah, DDS, MSc, FRCD(C),b and
P.A. Sikorski, DDS, MSc, FRCD(C),’ Toronto, Ontario, Canada

MCGILL UNIVERSITY AND UNIVERSITY OF TORONTO

The purpose of this study was to provide an objective analysis of the radiographic characteristics of
histiocytosis X as it affects the jaws. The original radiographs of 29 cases were reviewed
independently and objectively by three observers. The results suggest that seven characteristics,
either alone or in combination, are useful in the identification of histiocytosis X. These include the
appearance of solitary “intraosseous” lesions, the multiplicity of “alveolar bone” lesions, the
“scooped-out” effect in the alveolar process, the well-defined periphery, sclerosis in the alveolar
bone lesions, periosteal new bone formation, and slight root resorption.
(ORALSURGORALMEDORAL PATHOL1992;74:230-6)

H istiocytosis X is a term introduced by Lichten-

stein in 1953r for a group of diseasesthat produce
three syndromes with similar clinical and histopatho-
logic features: eosinophilic granuloma or chronic lo-
calized histiocytosis X, Hand-Schtiller-Christian dis-
ease or chronic disseminated histiocytosis X, and
Letterer-Siwe diseaseor acute disseminated histiocy-
tosis X. Other recent terminology for this group of
disorders includes idiopathic histiocytosis and Langer-
hans’ cell disease.‘,*
Although the concept that these three conditions
represent different manifestations of a single entity
has not been universally accepted,3-6most authors
now consider histiocytosis X as a spectrum of di-
seases.7-12 Not all patients can be accurately classified
under one of the three forms.‘2“4
The radiographic appearanceof the skeletal lesions
of histiocytosis X has been well documented, partic-
ularly in the skull, long bones, pelvis, and verte-
brae.5s15-23 The radiographic interpretation of histio-
cytosis X in the jaws can be difficult, as numerous
conditions can resemble histiocytosis X in radio-
aAssistant Professor, Department of Oral Radiology, McGill Uni-
versity.
bAssociate Professor, Department of Oral Radiology, University of
Toronto.
CAssistant Professor, Department of Oral Radiology, University of
Toronto.
7/16/37834
230
graphic appearance.5,8,17-19, 22-34Surprisingly, no de-
tailed study confined to the radiographic manifesta-
tions of histiocytosis X in the jaws could be found
in the English-language literature. Most publica-
tions on histiocytosis X in the jaws are case re-
ports. 13,24,29, 32,35-40
Holst et ale41reviewed 24 published cases,reported
3 new casesof eosinophilic granuloma of the jaws, and
gave a brief description of the radiographic appear-
ance of the lesions. These lesions were usually round,
oval, or irregular, resembled cysts, were occasionally
associatedwith a periosteal reaction, and were rarely
surrounded by bone sclerosis.
Rapidis et als4*analyzed 50 casesin the maxillofa-
cial region (43 casesfrom the English-language liter-
ature plus 7 of their own cases),but the radiographic
appearance of the osseouslesions was not discussed.
Hartmanr4 reviewed all aspectsof 114 casesof histi-
ocytosis X with oral involvement. The radiographic
description was a general overview of the appearance
of the lesions that affected the alveolar bone, which
were well defined and were capable of displacing
teeth. With severe bone destruction, the teeth ap-
peared to be “floating in air.” Artzi et a1.43reviewed
the periodontal manifestations in 28 published cases
of histiocytosis X with adult onset but did not describe
the radiographic appearance of the lesions.
To obtain meaningful characteristics to be used in
the identification of histiocytosis X would require an
Volume 74 Radiology for histiocytosis X 231
Number 2

objective analysis of a significant number of casesthat Table I. Location of the lesions in the jaws
included the original radiographs. The purpose of this No. of Percentage
study was to identify and determine the frequency of Site cases (%i
specific radiographic characteristics of histiocytosis X
of the jaws to facilitate the radiographic identification Mandible + ramus 28/29* 96.6
of this disease. Maxilla 9129 31
Mandible + ramus without 20129 69
MATERIAL AND METHODS maxillary involvement
Maxilla without mandibular l/29 3.4
Thirty-six casesof histologically proven osseousle- involvement
sions of histiocytosis X that affected the jaws were se- Maxilla + mandible together 8129 27.6
lected from the files of the Department of Radiology Ramus only 7/29 24.1
Intraosseouslesions 14129 48.3
of the Faculty of Dentistry of the University of Tor- Alveolar bone lesions 15129 51.7
onto. None of the caseswas diagnosed as Letterer- Alveolar bone-multiple 13/15t 86.7
Siwe disease.Seven caseswere excluded becausethe lesions
radiographs were of poor quality or provided insuffi- Mandible-bilateral lesions 11/15t 73.3
cient information to analyze the lesions. The other 29 *29 = total numberof cases.
radiographically well-documented cases were used. t I5 = numberof alveolarbonelesions.
There were 23 males and 4 females (sex was unknown
in 2 cases)with an age range from 2 to 45 years (mean eluded. The maxilla contained lesions in 9 cases
age, 17.3). Only the films taken at the time of the di- (3 I%), but only 1 case involved the maxilla without
agnosisof the diseasewere included. These films con- lesions in the mandible. Both maxilla and mandible
sisted of panoramic, anteroposterior, lateral, and were involved in 8 cases(27.6%).
submentovertex skull views, oblique-lateral views of There were 14 solitary intraosseouslesions: 1 in the
the jaws, and intra oral films. maxilla, 6 in the mandible below the inferior alveolar
All caseswere studied separately by three observ- nerve canal, and 7 in the ramus (1 of which was in the
ers trained in oral radiology. The characteristics of condyle). Five of the 7 lesions of the ramus were lo-
each case were recorded on a checklist designed to cated in the lateral aspect of the ramus. Approxi-
obtain a simple “yes” or “no” decision. This analysis mately one half of the lesions occurred in the alveolar
included the location, shape, size, periphery, and in- process(15 cases,5 1.7%), whereas the other 14 cases
ternal structure of the lesion, the presence of peri- (48.3%) were classified as intraosseous. It is of inter-
osteal new bone or expansion, and the effect on teeth est to note that when two or more lesions were present,
and adjacent structures. they were always located in the alveolar process.
For the purpose of comparison, lesions were sepa- Alveolar bone lesions were present in at least 2
rated on the basis of their location: maxilla, mandible quadrants in 13 of the 15 cases(86.7%), and in 11 of
(excluding ramus), and ramus, or any combination of these cases the mandible had bilateral involvement
the three. The lesions were further divided into those (Fig. 1). In the seven cases in which the epicenter
that had originated in the alveolar process (alveolar could be determined, the lesions appeared to have
bone lesions), and those surrounded by bone but that started in the apical region of the teeth, and in nine
had originated outside the abeolar process (in- casesthe lesions originated from the area of root fur-
traosseous lesions). This was accomplished by iden- cation of the teeth. In five casesboth the region of the
tifying the center of the lesion and assuming equal furcation and the apex were involved at the sametime.
growth in each direction. None of the lesions appeared to have started from the
A specific characteristic was considered to be summit of the alveolar bone as is seenin periodontal
present if at least two observers felt confident of its disease.All the lesions originated from the posterior
presence.The results were expressedas a percentage regions of the jaws (posterior to the canine tooth). The
of the total number of casesin which the observation anterior teeth (canine and incisors) were involved in
could be made. Further statistical analysis was not three casesby extension of very large alveolar bone
possible because of the absenceof a control group. lesions that originated in the posterior regions. In 14
of 15 cases,lesions that affected the alveolar process
RESULTS causedbone destruction that remained superior to the
The locations of the lesions are shown in Table I. inferior alveolar canal.
Of the 29 cases, the mandible and ramus were The assessmentof the shape and periphery of the
affected in 28 cases(96.6%) and in 20 cases(69%) if intraosseouslesions revealed that 13 (92.9%) were el-
simultaneous involvement of the maxilla was ex- liptical or circular in shape, 9 (64.3%) had well-
232 Dagenais, Pharoah, and Sikorski ORAL SURG ORAL MOD ORAL P?\THOL
August 1992

Fig. 1. Panoramic radiograph with bilateral alveolar bone-type lesions. Note the lesions have extended an-
teriorly to involve the cuspid regions.

Table II. Frequency of occurrence of characteristics

of histiocytosis X of the jaws
No. of Percentage
Characteristic cases (%o)

Well-defined intraosseouslesions 9114s 64.3

Well-defined alveolar bone lesions 8/15? 53.3
Scooped-out effect 8/15 53.3
Sclerosis IO/l5 66.7
Periosteal new bone-total 14/29$ 48.3
Periosteal new bone-intraosseous 12114 85.7
Root resorption 8115 53.3
*14 = total number of intraosseous lesions.
t15 = total number of alveolar bone lesions.
$29 = total number of casesstudied.

Fig. 2. Periapical film shows well-defined alveolar-type

lesion with a scooped-out effect (arrows).
defined margins (Table II), and 5 (35.7%) had ill-de-
fined or invasive margins. In the alveolar process,
multiple lesions with different shapes were sometimes
present in the same patient. In 53.3% of the cases that
involved the alveolar process, the lesions were de-
scribed as having a “scooped-out” shape (Figs. 2, 3,
and 4). A scooped-out appearance was defined as a
circular or oval-shaped area of bone destruction in the
alveolar process in which at least a portion of the crest
of the alveolar ridge is still intact (Fig. 4). The same
percentage (53.3%) of alveolar bone lesions had well-
defined margins. A margin was termed well defined
when the observer could easily outline the extent of
the lesion. A sclerotic border or some degree of cor-
tication of the periphery of the lesions was noted in 10
cases (66.7%) that affected the alveolar process, but
was absent in the intraosseous lesions.
The presence of calcified internal structure was
found in only two cases. Expansion of the bone was
noted in two cases that were located in the condyle and
the coronoid process. Periosteal new bone formation
(Fig. 5) was found in 14 of the 29 cases (48.3%);
however, this was a frequent finding in intraosseous
lesions of the mandible (12 of 14 cases, 85.7%). The
amount of periosteal new bone was judged to be
prominent in 10 cases (71.4%).
Deciduous molars and permanent premolars and
molars were the most frequently affected teeth. The
entire lamina dura or portions of it were absent in all
the cases that affected the alveolar process. In-
traosseous lesions that reached the apices of teeth also
caused loss of the lamina dura. Lesions adjacent to
developing teeth resulted in destruction of the follic-
ular cortex, displacement of follicles, and displace-
ment of tooth buds within their follicles. Very mild
root resorption was a frequent observation in alveolar
bone lesions (53.3%). Teeth totally bereft of osseous
support (floating in air) were observed in only five
cases.
A thorough study of the effect of lesions on the
mandibular canal, floor of the antrum, and floor of the
Volume 74
Number 2
Fig. 3. Panoramicradiographshowsmultiple well-definedalveolar-typelesions.Note scooped-out
the mandible (arrow).
nasal fossaeproved to be difficult becauseof the lim-
itations of the radiographic surveys. However, in
well-documented cases the cortical boundaries of
these structures were destroyed.
DISCUSSION
In this study the following seven radiographic
characteristics occurred frequently with histiocytosis
X of the jaws (Tables I and II).
1. Solitary intraosseous lesions (outside the alve-
olar process) (Figs. 5 and 6). Contrary to the
opinion that most lesions of the jaws involve the
alveolar bone alone,*, 9,15,19,33,44-46
in this study
nearly one half (14 of 29 cases) of the lesions
were classified as having started outside the al-
veolar process(intraosseous). Unlike the alveo-
lar bone lesions, intraosseous lesions were al-
ways solitary and the majority had a circular or
elliptical shape.The condylar lesion in this study
is only the third lesion reported in that loca-
tion.47>48It is of note that most of the lesions of
the ramus were located in the lateral aspect of
the bone.
2. Multiplicity of “alveolar bone” lesions. Our
results indicate that more than one quadrant
was frequently affected when the lesions in-
volved the alveolar bone (Fig. 3). These results
are in accord with the findings of Blevins et a1.8
3. Well-dejined periphery. The periphery of the
lesions of histiocytosis X in the jaws was consid-
ered to be well defined but uncorticated by most
authors.14T25126,44,46149)5o The term punched-
out has often been used to describe the lesions
in other bones5, 12,15,16,22,51-54Our data were
in agreement since the majority of the lesions of
the alveolar bone had well-defined margins
(Figs. 2 and 3). However, 35.7% of other
intraosseous lesions had ill-defined or invasive
Radiology for histiocytosis X 233
effectin
A
B
C
Fig. 4. Diagramusedasguideto classifyshapeof alveo-
lar lesions.Fig. C representsthe scooped-outappearance.
margins in contrast to the alveolar bone lesions
(Fig. 6).
4. Scooped-out shape. It is significant that a
scooped-outshapewas observedin 53.3% of the
alveolar bone cases,(Figs. 2,3 and 4). This par-
ticular appearanceoccurs becausethe bone de-
struction starts below the crest of the alveolar
process.In 9 of the 15 alveolar bone cases,it was
determined that the area of destruction started
at the level of the furcation or halfway down the
root of the tooth. Usually a portion of the supe-
rior aspect of the crest of the alveolar bone is
maintained at the mesial and distal margins of
the area of destruction and produces a scooped-
out appearance. Zuendel et a1.55reported one
casewith this characteristic. It is possible that,
with progression of the destruction, the bone
that persisted at the superior aspect of the crest
is destroyed and this characteristic is lost. This
234 Dagenais, Pharoah, and Sikorski
Fig. 5. Cropped panoramic radiograph shows ill-defined
intraosseouslesion with associatedperiosteal new bone for-
mation (arrow).
form of bone destruction has not been observed
with periodontal disease and thus may be useful
in the differentiation of lesions of histiocytosis
5. Bone sclerosis. Bone sclerosis is not considered
a feature of histiocytosis X in other bones and
has been attributed to healing.‘5, 18,22,23,51,53
Sclerosis is a common observation in inflamma-
tory lesions of the jaws and the fact that it ap-
pears frequently in the alveolar bone lesions
might be explained by a communication of the
lesions with the oral cavity that results in a su-
perimposed infection. In support of this theory
is our observation that the intraosseous lesions
that did not communicate with the oral cavity
did not present any evidence of cortication or
sclerosis. Sedan0 et a1.i9 were the only authors
to describe sclerosis associated with lesions of
the jaws.
6. Periosteal new bone. Periosteal new bone for-
mation was observed in a significant number of
cases (12 of 14) that were classified as in-
traosseous lesions. Although not frequently re-
ported in the jaws,42, 46,47,56,57 the high fre-
quency of occurrence in our study indicates that
it may be a good characteristic to use in the
identification of histiocytosis X. The identifica-
tion of the presence of this thin layer of bone is
highly dependent on the projections available
for study, and our data may not reflect the ac-
ORAL SURG ORAL MED ORAL PATHOL
August 1992
Fig. 6. Cropped panoramic radiograph shows in-
traosseous lesion with ill-defined periphery in ramus of
mandible (arrow).
tual occurrence. Many of the alveolar bone le-
sions were not examined with occlusal radio-
graphs that would have allowed for the evalua-
tion of the buccal and lingual aspects for
periosteal new bone.
7. Root resorption. Root resorption, as reported by
Dombowski,25 was a common observation in this
series. However, the resorption associated with
lesions of histiocytosis X was always very slight.
Except for the large number of intraosseous lesions,
the remaining findings about the location of the
lesions were in agreement with the observations of
most authors.*
The effect of histiocytosis X on teeth also agreed
with findings previously reported.? Displacement of
erupted teeth and follicles was the most common ra-
diographic finding for Blevins et a1.,8 but it did not
occur very frequently in the 29 cases reported here.
This can be explained by the fact that very few cases
reviewed involved unerupted teeth. The fact that
many of the lesions observed were small would
account for the low number classified with floating
teeth as compared with the frequent observations in
the 1iterature.S
To our knowledge, this is the first attempt to iden-
tify and quantify the radiographic characteristics of
*8, 14, 15, 17, 19, 26, 33, 46, 58
t8, 14, 15, 17, 21, 27, 33, 46, 47, 50, 52, 53, 59
$14, 15, 17, 18, 42, 44, 46, 47, 50, 53, 60
Volume 74
Number 2
histiocytosis X of the jaws. Since radiographic char-
acteristics are difficult to measure, we employed an
objective analysis carried out by three radiologists.
The quality, number, and types of radiographs used
in each casedid limit the number of observations that
could be made. An improvement in the quantity and
quality of these data would probably result if oral ra-
diologists were routinely given the opportunity to ex-
amine all histiocytosis X patients.
In summary, this study demonstrated that the fol-
lowing radiographic characteristics are useful in the
diagnosis of histiocytosis X of the jaws: the appear-
ance of round or oval solitary intraosseouslesions as-
sociated with periosteal new bone formation; the
presence of multiple lesions of the alveolar process
that have a well-defined periphery; a scooped-out ef-
fect; sclerosis; and mild root resorption.
The significance of the establishment of reliable
radiographic characteristics lies in the fact that
biopsy and histopathologic examination do not always
reveal the true nature of this disease.The formation
of a radiologic diagnosis will aid in the subsequent
histopathologic examination and will allow the use of
nuclear medicine in the search for multiple lesions.
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Reprint requests:
M. J. Pharoah, DDS, MSc, FRCDtC)
University of Toronto
Faculty of Dentistry
124 Edward St.
Toronto, Ontario, Canada M5G lG6