a pedicle in the cervical esophagus to the gastroesophageal junction, with its distal tip prolapsed into the ... bulging into the superomedial aspect of the gastric.
Leiomyosarcoma
of the
Radiographic
Marc James Linda Peter James Leslie
OBJECTIVE.
S. Levine1’2 L. Buck1 Pantongra9-BroWn1 C. Buetow’ R. HaIIman3 H. Sobin3
tumors
that
Leiomyosarcomas
have
of this study
been
AND
Institute
of
described
was to evaluate
MATERIALS Forces
Findings
only
findings
RESULTS.
the
rare
of esophageal
with dysphagia.
lesions
in six patients,
in two, and infiltrative
lesions
all had large
lesions
lesions components,
appeared
archives
objective lesion.
of the Armed
leiomyosarcomas. The average
had dysphagia in five patients.
the intraluminal
The
of this unusual
Clinical
retrospectively.
presented
intraluminal
smooth-muscle
literature.
findings
phagia was 6.7 months, but five patients radiographs revealed a mediastinal mass exophytic
malignant
of the radiology
I 0 cases
reviewed
All but one patient
are
in the radiology
A search
revealed were
esophagus
and radiographic
METHODS.
of Pathology
and radiographic
in 10 Patients
anecdotally
the clinical
Esophagus:
and three
as a polypoid
duration
of the dys-
for 3 or fewer months. Frontal chest Barium studies revealed intramural lesions
contained
expansile
in two. The intramural
ulceration
mass
and
or tracking.
the other,
One of
as a smooth
expansile sausage-shaped mass mimicking a fibrovascular polyp. CI’ revealed a mass involving the esophagus in five patients: three of these patients had heterogeneous lesions containing large exophytic components, central areas of low density, and extraluminal within the tumor. In two patients, MR imaging revealed large masses skeletal
muscle
on Tl-weighted
CONCLUSION. radiographic findings tract.
Esophageal
and are often
S Received December February 19, 1996.
26, 1995; accepted
after revision
of Radiologic Institute
Pathology,
of Pathology, Washington,
Armed Forces
DC 20306.
2Department of Radiology, Hospital of the University of Pennsylvania, 3400 Spruce St., Philadelphia, PA 19104. Address correspondence to M. S. Levine. 3Department of Hepatic and Gastrointestinal Pathology, Armed Forces Institute of Pathology, Washington, DC
20306.
ince
Roentgen
AJR:167, July 1996
description
a better
in
nized
as
fewer
than
ageal
tumors
1%
of all
[2-5].
Because
by
metastases,
malignant
slow
it has
esoph-
this tumor
growth
a better
and
prognosis
is late
carcinoma [3, 6, 7]. In the leiomyosarcomas have
been
into
classified
polypoid
and
infiltra-
tive types on the basis of the gross pathologic findings [4, 8-10]. In various anecdotal revealed masses lesions
reports, polypoid or, less with
barium
studies
expansile commonly,
irregular
have
intraluminal infiltrative
luminal
In several
tral
areas
of low
density
prognosis
than
images. leiomyosarcomas have in the gastrointestinal
squamous
cell carcinomas
narrowing
cases,
19, 10,
Recently,
however,
leiomyosarcoma intramural mass
we
saw
an esophageal
that appeared as a giant on barium study and as a
cavitated lesion on CT. This review the radiology archives
prompted us to of the Armed
Forces Institute of Pathology radiographic findings of this
to reassess the unusual tumor.
than
squamous cell past, esophageal
CT has
thickening of the esophageal or a soft-tissue mass with or without Ray Society
esophageal elsewhere
have
shown
0361-803X/96/1671-27
on T2-weighted
suggests that of leiomyosarcomas
cure.
to surgical
its original
and hyperintense
1902 [1], leiomyosarcoma of the esophagus has been recoga rare lesion that constitutes
[3, 5, 8, 10-14].
AJR 1996;167:27-32
© American
leiomyosarcomas amenable
characterized
The opinions and assertions contained herein are the pri vate views of the authors and are not to be construed as reflecting the views of the Departments of Defense, Army, Navy, or Air Force.
images
Our experience similar to those
gas or contrast material that were isointense with
14,
Materials
A search of the radiology Forces Institute of Pathology
archives
of the Armed
revealed
eight cases
esophageal
contributed
leiomyosarcomas
from
of
I 978
to 1993 and two cases contributed before 1978. These 10 cases formed our study group. Medical records were reviewed to determine the clinical presentation. All 10 patients had barium studies that were
reviewed
radiographic
wall
had
cen-
MR
15].
and Methods
retrospectively features
of
chest radiographs.
studies,
and
seven
these five
to
determine
tumors.
Six
the patients
had CT scans, two had
had
tions, which were also reviewed.
endoscopic
examina-
Levine
Seven mies,
patients two
lesions
without
underwent
Institute coma
adjacent
pathologic
localized
resection. and one unresectable tumor that structures. In all patients. the
of an
diagnosis
of Pathology (Fig.
of
at the
was esophageal
Armed
Forces
letomyosar-
I).
black, and one was Hispanic. Nine patients presented with dysphagia. The average dura-
Results
esophagogastrecto-
enucleation
Clinical
Findings
The
esophageal
debulking
had invaded final
underwent
underwent
et al.
average
esophageal old
(range,
patients were Five patients women.
age
of our
leiomyosarcomas
10 patients
with
was
61 years
All
but
44-81
years).
between were
50 and 70 years old. men and five were
Six patients
were
white,
three
two
were
tion ofdysphagia 30
months),
was 6.7 months
However, for 3 or
dysphagia
five fewer
(range,
I-
patients months.
had The
remaining patient had signs of gastrointestinal bleeding (melena and anemia) without Other
dysphagia.
included
weight
tric pain
in three,
in two, anorexia emesis
presenting
findings
loss in five patients,
in one,
epigas-
chest
pain
in two,
fatigue
in one,
early
satiety
in one,
and fever
in one.
Chest Film Findings
Frontal (one),
chest
left-sided
mediastinal
Barium
leiomyosarcoma. Photomicrograph of histologic section shows mulwith at leastthree mitoses (arrows) on this high-power view. Findings tumor of esophagus. )H and E, original magnificationx400)
studies lesions
intraluminal
in two (Fig.
3A).
revealed
predominantly
in six patients,
predomi-
lesions
and
in two,
infil-
lesions in two. Seven patients had in the distal esophagus and two had in the midesophagus. The remaining had a lesion extending cervical esophagus through
troesophageal dus.
(one)
in the lower
Studies
trative lesions lesions
Fig. 1.-Histopathologic findings of esophageal tiple spindle-shaped cells arranged in fascicles were compatible with malignant smooth-muscle
right-sided bilateral
thorax in three 2A) and a right-sided mediastinal
(Fig.
intramural
patient lower
and
in the midthorax
Barium nantly
revealed
masses
patients
mass
films (one),
The
junction average
9.8 cm (range,
into
length 5-25
from the
the gastric
of the
lesions
the gasfunwas
cm).
Fig. 2.-Esophageal leiomyosarcoma in 59-year-old man. A, Frontal
chest
radiograph
shows
mediastinal mass (arrows) extending into both sides of lower thorax. B, Barium study shows large smooth slightly
distal
lobulated
intramural
mass in
Note exophytic component of tumor (arrows) extending into mediastinum. A 28
esophagus.
B AJR:167, July 1996
Imaging
of Leiomyosarcoma
of the Esophagus
Fig. 3.-Esophageal
leiomyosarcoma in 69-year-old man. chest radiograph shows mass (arrows) extending into chest from right mediastinum. study shows smooth intramural mass (large black arrows) with large exophytic component arrows) communicating with esophageal lumen. C, In another projection of same study as B, lesion is shown en face, with splitting of barium around
A, Frontal B, Barium
In six patients. intramural masses
formed right angles with the adjacent
linear
obtuse angles wall. The
with
its
f’undus
distal
tip
(Fig.
radiographs
revealed
bulging
into air
gastric
bubble
a
(Fig.
three ation,
appeared rowing
including
was
(Fig.
round, their
of
a 2-cm
in four,
intraluminal
smaller
a 6-cm
ulcer,
the tumor the lesions were
they
were
than
the
spheroid
with
along the long all six patients.
component
extended
ulcer-
within
patients.
long axes oriented the esophagus. In
of
exophytic
the
lesion
component
axis the that
6).
expansile extended esophagus
mass The
in the
other
distal
patient
a pedicle
esophagus
had
sausage-shaped from
overhanging
a smooth mass
in
to the gastroesophageal
the
This
however,
phytic
components
the
had
fibrovas-
lesions
that
areas of luminal ulceration and
narshelf-
that
carcinomas
cases,
of the
lesion
of a giant
borders
cell
squamous
(Fig.
resembled 8). In both
had large exointo the medi-
lesions
extending
astinum. contained
In one of these, the lesion an irregular 3-cm track that com-
municated
with
the esophageal
lumen.
Cl’ revealed
a mass
CT Findings
In five patients,
of the two patients with intraluminal had an irregular polypoid expansile
intraluminal
like
mass
aspect
infiltrative
as irregular with extensive
gastric
was
into the mediastinum.
One lesions (Fig.
ulcer.
visualized
In two
and
contained
linear track (Figs. 3B, 4, and of these patients. extraluminal
faintly
5A).
lesions
black
abdominal
soft-tissue
7B).
17). had
the
case,
the superomedial
cularpolyp [16, Two patients
the
(small
into
In this
lesions had a smooth contour in four patients and a somewhat irregular contour in two. In patients,
track
prolapsed
7A).
features
gas
AJR:167, July 1996
contains
mass.
located outside the and their borders
or slightly esophageal
Note that lesion
the radiographic
and a 3-cm 5A). In one
leiomyosarcoma in 58-year-old woman. Barium study shows large, somewhat irregular intramural mass (large black arrows) containing giant central ulcer (small black arrows). Note narrow neck of ulcer (white arrow) communicating with esophageal lumen.
arrows).
the lesions had features of (Figs. 2B. 3B, 3C. 4. and
5A); their centers were lumen of the esophagus,
Fig. 4.-Esophageal
(white
that cervical
junction,
involv-
ing the esophagus: three of these heterogeneous lesions containing
patients large
phytic
of low den-
components,
central
sity, and extraluminal within
the
three
patients,
the wall
tumor
(Fig,
the lesions
of the
areas
gas or contrast
gastric
SB).
In two
extended fundus.
had exo-
material of these
distally
into
In the fourth
29
Levine
patient, the homogeneous
lesion appeared as a relatively soft-tissue mass involving the
esophagus. CT,
In the remaining
the
lesion
esophageal
mass
was thought This
appeared
as an
imaged
junction
MR Imaging
Findings
density
fibrovascular
also extended
esophageal
on
intraluminal
of homogeneous
to be a giant
lesion
patient
through
that polyp.
one two
In two patients.
MR imaging
(Fig.
SC)
images
and
(Fig.
contained extraluminal Endoscopic
Of the endoscopy. Endoscopic
hyperintense SD).
fundus.
a focal
area
gas within
patient,
of signal
isointense images
due
patients.
The
erroneous
diagnoses.
carcinoma
and
one
one
four and
in patients, two had
of squamous
of Barrett’s
cell
biopsy
a leiomyosarcoma
cell
esophagus. confirmed in only one
patients.
of the
to
the tumor.
Findings
seven patients who underwent lesions were visualized in six. biopsy specimens were obtained
smooth-muscle
less
frequently
tumors than
or small
that occur
leiomyosarcomas
spindle mitotic
bowel
[2-5].
These
the number
lesions
power
has
field,
from Some
can
figures
in each
authors
have
pericardium, diaphragms, and they metastasize hematoge-
biologic behavior of the only sign of malignancy
nously to the 101. Because
liver, lungs, esophageal
usually
more
slowly
than
squamous
be difficult
but other
lungs, pleura, stomach, or
grow
consist of pleo-
of mitotic
the reproducibility mitotic counts
appears
occasionally
leiomyomas by pathoauthors have relied on
tend to be low-grade malignant tumors that eventually spread by direct extension to the
and bones [5, 7, leiomyosarcomas
15].
cells with increased numfigures [5, 10] (Fig. 1).
leiomyosarcomas
much
sur-
of an esoph-
leiomyosarcomas of interlacing whorls
of the
are
with 5-year 35% [5,
progression
ageal leiomyosarcoma been documented [151. Esophageal pathologically
leiomyosarco-
prognosis. approach
rapid
to differentiate logic criteria.
esophagus
esophageal
Nevertheless,
However,
rare
stomach
carcinomas,
mas have a better vival rates that
morphic bers of
Leiomyosarcomas
the lesion void
six
Discussion
examinations
on T2-weighted
In one
these revealed
patient. Of the remaining had nonspecific findings
of seven
revealed large masses that were with skeletal muscle on TI-weighted
of
specimens
Thus, endoscopy before surgery an esophageal leiomyosarcoma
the gastro-
into the proximal
in five
et al.
[7,
high-
doubted
and prognostic value of 13, 18]. Ultimately, the
to be histologically
lesion may be the in a tumor that benign.
Most esophageal leiomyosarcomas thought to arise de novo rather than from
are pre-
Fig. 5.-Esophageal Ieiomyosarcoma in 67-year-old woman. A, Barium study shows giant intramural mass (large arrows) with bulky exophytic component in mediastinum (open arrows). Note relatively small central ulcer (small arrow) and extraluminal gas (arrowheads) faintly seen within lesion. B, CT scan shows heterogeneous mass (white arrows) in left mediastinum with central areas of low density. Note extraluminal collections of gas (solid black arrows) within ageal lumen (open black arrow). C, Ti-weighted (TRITE, 674/12) MR image shows mass (straightarrows)
lesion that are separate in left mediastinum.
from esophNote that mass
is isointense with skeletal muscle. D, T2-weighted (2697/80) MR image shows that lesion (straight arrows) now is markedly hyperintense relative to skeletal muscle. Note that both C and D reveal focal area of signal void (curved arrows) caused by extraluminal
gas within
tumor.
A
30
AJR:167, July 1996
Imaging
Fig. 6.-Esophageal leiomyosarcoma in 60-year-old man. Barium study shows irregular polypoid intraluminal mass (arrows) expanding lumen of distal esophagus.
of Leiomyosarcoma
Fig. 7.-Esophageal
of the Esophagus
leiomyosarcoma
in 44-year.old woman.
A, Three views from barium study show smooth expansile sausage-shaped mass extending from pedicle in cervical esophagus (white arrows) to gastroesophageal junction, with distal tip (black arrows) prolapsed into gastric fundus. B, Abdominal radiograph shows soft-tissue mass (arrow) bulging into superomedial aspect of gastric fundus.
propria
muscularis
muscle
is composed
in this portion
9]. Occasionally, can develop from
however, from
smooth
in the walls
esophagus
Esophageal found
common occurring
more
presenting in 75-85%
existing leiomyomas are most conimonly
proximal
(arrows).
110. 191. These found in the middle
tal third
of the thoracic
AJR:167,
July
1996
borders
esophagus
lesions or dis-
because
the
on chest
usually
common
on
[3,4,
in men than is the
clinical of reported
most
complaint, cases [7, 91
frontal
chest
patients (Figs. myosarcomas
[5, 8, 9,
radiographs this finding
was present
radiographs
in
50%
the differential in patients
diagnosis of mediastinal with dysphagia. Rarely,
tumors
contain
can
of
2A and 3A). Esophageal leioshould therefore be included in
dense
masses these
calcification
seen
been
cases
and as infiltrative in 40% [9, 10]. Polylesions have appeared on barium studies
or absent
20. 211. When time
50%
and shelflike
mediastinum
in the
have
be minimal
growth
ulceration,
in
can
by a mass
can
the
esophagus with irregular luminal narrowing, extensive
of vessels
leiomyosarcomas
be recognized
on chest radiographs [22J. In the past, esophageal
it has often
in 67-year-old lesion in distal
sometimes
farther into the medicarcinomas. As a
and in 90% of our study population. However, as one of our cases illustrates, dysphagia
lumen
leiomyosarcoma shows infiltrating
esophageal
or
are
if the tumor
predominantly exophytic pattern without significant encroachment
Fig. 8.-Esophageal man. Barium study
result,
mucosae
patients
[4, 5, 9]. Dysphagia
in women
they tend to grow much astinum than esophageal
1 1, 20]. In our study,
or elderly
7] and are slightly
[4, 5,
[6].
leiomyosarcomas
in middle-aged
smooth
leiomyosarcomas
the muscularis
muscle
the proximal
of
of the esophagus
fewer toms include
been
of growth on the
dysphagia
present
of diagnosis
does
because
of the
loss, chest
pain,
Because often have
esophageal an exophytic
slow
of sympfindings
regurgitation,
due to comprestree by tumor [5-
71. Although rare, gastrointestinal can occur if the tumor is ulcerated
bleeding [9].
leiomyosarcomas pattern of growth,
poid
as expansile at
[5]. Nevertheless, dysphagia for 3 or
months; thus, the duration is variable. Other presenting weight
occur,
for a year or more
of these tumors of our patients had
and respiratory symptoms sion of the tracheobronchial
has a
ically
can
dysphagia versely, irregular
leiomyosarcomas
as polypoid
intraluminal become
masses
enormous
in 60%
of
that paradoxbefore
causing
[3, 5, 8, 10-14) (Fig. 6). Coninfiltrative lesions have appeared as areas of luminal narrowing with typi-
cal features nomas
classified
of advanced
squamous
cell carci-
[3, 8, 10] (Fig.
8). These
infiltrative
lesions have classically been associated with early dysphagia. Patients with these lesions have
a poorer
prognosis
polypoid lesions [7, 81. In our study, however, leiomyosarcomas studies as large
(60%) intramural
than most appeared masses,
patients
with
esophageal on barium often con-
31
Levine
taming areas of ulceration or tracking (Figs. 2B, 3B, 3C, 4, and 5A). The tumors also had large
exophytic
from
the
esophagus
Therefore, ageal
components into
features
graphic
stomach
that esoph-
the same
as leiomyosarcomas bowel
and
other
[23, 24]. benign
can also appear masses,
away
mediastinum.
suggests have
and small
leiomyomas mural
the
our experience
leiomyosarcomas
tumors
extending
accurate patients
and are
the diagnosis
reports,
esophageal
have
been
by thickening
of leioleiomyo-
manifested
on CT scans
of the esophageal
soft-tissue
mass
that
wall
characteristic neous lesion
sometimes
contains
finding on CT was a heterogecontaining large exophytic com-
central
extraluminal
areas
gas
low
of
or contrast
density,
material
and within
the tumor (Fig. SB). The latter finding presumably occurred as a result of communication of the lesion with the esophageal lumen and subsequent cavitation of the necrotic portions
of the tumor.
Similar
CT findings
been reported with leiomyosarcomas stomach and small bowel [26-28],
have
of the indicating
that these tumors have identical gross pathologic features regardless of their location in the gastrointestinal tract. The findings on MR imaging have recently been ageal
described in a patient leiomyosarcoma [29].
with an esophThe lesion was
manifested by a mass that was isointense with skeletal muscle on TI -weighted images and hyperintense on T2-weighted images. In our study,
two
patients
findings (Figs. Recently,
had
from
ageal wall recognized masses channels
the
imaging
of the esoph-
tumors can also be as hypervascular
vessels, dilated vascular lakes, and early venous
However,
leiomyosarcomas.
we have
the tumor
32
Endoscopic
can be helpful is ulcerated
no experi-
biopsy
if the mucosa [30].
The
specimens
of
their
slow
Esophageal
presin
Med
1982;82:l
superficial precludes
histologic diagnosis with this tumor.
An esophagectomy
in
growth
and
[4-7, 9]. a palliative Rarely,
has been achieved
vival
nonsurgical esophageal features
our
experience
leiomyosarcomas
and often
ation or tracking.
that
CT usually
areas
of ulcer-
revealed
heteroge-
lesions with central areas of low density and extraluminal gas or contrast material within neous
the tumor. Finally, MR masses that were isointense cle on Tl-weighted T2-weighted
It is important
of the radiographic myosarcomas,
imaging revealed with skeletal musand hyperintense
images
images.
speci-
overlying
If the overlying
13. Gaede
features as these
than esophageal often amenable to surgical
esophageal
cases
in the archives
logic
Pathology
Pathology.
of
We
pathologists
of the Department the
thank
whose
to the AFIP
Armed
all
the
contributions have
made
of Radio-
Forces
Institute
of
radiologists
and
the esophagus.
tumors ofthe
WT. Primary
sarcoma
20.
Pennes
AiR
3. Rainer WG, Brus esophagus. Surgery
R. Leiomyosarcoma
Sarcoma
AE.
Lyons
Leiomyosarcoma
of the esophagus.
Mil
AS,
comas
Shimazu
of
of the e.sophagus.
Imaging
growing
report and review 1995; 20:15-19
uterus.
Hum
of the mitotic count of smooth muscle
1976:7:451-454
Pathol
M. The radiological
Leiomyosarcoma
approach
of the
Garlock
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JH.
Leiomyosarcoma
of the
Surgers’ 1951:29:281-287 H. Leiomyosarcoma
Zornoza
ofthe
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MS. Benign
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In: Levine Philadelphia:
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Clark RA, Alexander ES. Computed of gastrointestinal leiomyosarcoma.
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29. Ohnishi D.
Med 1988;
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TC,
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1, Yoshioka H, Ishida 0. MR imaging of
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14. Aimoto sarcoma
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on
of esophageal
DF. Unusual
agus: report of two cases, one associated with squamous cell carcinoma. J Thorac C’ardiovasc Surg 1978:75:740-746
to be aware
tumors
J
CA, Anti IM. Leiomyosarcoma Gastroenterologv 1968:54:271-274
IL, Hamilton
of leiomyosarcomas
contained
Radiol
12. Athanasoulis esophagus.
in
suggests
GS, Bode
N YState
Gastmintest
sur-
found elsewhere in the gastrointestinal tract. Barium studies most commonly revealed large intramural masses that had a marked exophytic component
esophagus.
have radiographic
to those
similar
is
[5, 7, 9].
candidates
In summary,
Y State
100-1 103
I I . Wolfel DA. Leiomyosarcoma AiR 1963:89:127-131
therapy
Rosenthal
N
the gastrointestinal tract.AJR 1971;l 13:159-170 9. Patel SR. Anandarao N. Leiomyosarcoma of the
late
prolonged
WL,
10. Balthazar El. Gastrointestinal leiomyosarcomaunusual sites: esophagus, colon, and porta hepatis.
Even if resection
by radiation
AS, Thelmo
leiomyosarcoma.
8. Berk RN, Scher
an many
or esophagogastrectomy
the surgery of choice metastases are present.
cases
ence with these imaging techniques. It is important to be aware of the limitations of endoscopy in diagnosing esophageal mens
patients.
WS.
leiomyosarcomas
layer
[14, 15]. These on angiography
[10].
MR
on endoscopic sonoghyperechoic masses that
muscular
with tumor or venous
drainage
similar
SC and SD). esophageal
have been characterized raphy by well-defined arise
confirmed the leiomyosarcoma
GO, Antler
In
[30].
metastases, even large esophageal leiomyosarcomas can be amenable to surgical resection.
or by a
areas of low density due to necrosis of tumor [9, 10, 14, 15]. In our study, however, the
ponents,
of seven
7. Franklin
false-negative
be obtained
can still be performed.
myosarcoma. In anecdotal sarcomas
suggest
however, may
of the biopsy
Because
as intra-
they tend to be smaller
one
of the
if ever ulcerated [25]. Therefore, large intramural lesions that have ulceration or should
only nature
Although
intact,
our study, endoscopy ence of an esophageal
mesenchymal
on radiographs
is
specimens
radio-
rarely
tracking
mucosa biopsy
et al.
EK, Sanoski
RA,
scopic diagnosis of a giant sarcoma. Am J Gastroenterol
Kozarek
RA.
Endo-
esophageal leiomyo1981:75:132-134
AJR:167, July 1996