Leiomyosarcoma of the Esophagus

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

esophagus.

JH.

Leiomyosarcoma

of the

Surgers’ 1951:29:281-287 H. Leiomyosarcoma

Zornoza

ofthe

oesophagus

BrJRadioll9785l:469-47l

J, Ordonez

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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.

tomography Gastrointest

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29. Ohnishi D.

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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