Chondrosarcomas arising from soft tissues are rare. Two different varieties are described, myxoid and mesenchymal. We have collected nine cases of the ...
CHONDROSARCOMA
OF
TWO
R.
CASADEI,
M.
RICCI,
P.
DIFFERENT
RUGGIERI,
From
THE
R.
Rizzoli
of chondrosarcoma
and histological Extraskeleta!
seen
differences
between
BIAGINI,
S.
Orthopaedic
chondrosarcomas
are
between
1904
less
common
than those in bone. Tumours arising from the soft tissues are not attached to bone, periosteum or cartilage (Stout and Verner 1953 ; Go!denberg, Cohen and Steinlauf 1967; Amir et a! 1985). They can be divided into two histological varieties : myxoid and mesenchymal. The aim
of this
paper
is to define
PATIENTS
these
AND
two
entities.
METHODS
At the Bone Tumour Centre of Bologna, 5 1 3 cases ofchondrosarcoma 1904 and 1988 and have been
the
radiographical!y and histologically. was the diagnosis of soft-tissue firmed (1 7%). Of these, five were
Only in nine patients chondrosarcoma conmyxoid and four were
BENASSI,
Institute,
P.
PICCI,
M.
CAMPANACCI
Bologna
Two different varieties are described, five myxoid and four mesenchymal,
and 1988.
the two varieties
much
TISSUES
SUB-GROUPS
Chondrosarcomas arising from soft tissues are rare. mesenchymal. We have collected nine cases of the tumour, of 513 cases
SOFT
We report
and discuss
their
Clinical
findings.
the principal surgical
clinical,
treatment
Clinical
myxoid and from a review radiographical
and prognosis.
details
are
reported
in Table
toms.
ranged
from
months (average, 12 months : 1 7 for mesenchyma! chondrosarcoma). Imaging. In myxoid chondrosarcoma were normal (case 3) or showed only
The
duration
of symptoms
myxoid,
three
to 24
nine
Rizzoli Institute, were seen between reviewed clinically,
Correspondence
©
should
be sent
Centre,
and Via
Head Di
of the
Barbiano Fig.
1
to Dr P. Picci.
1991 British Editorial Society ofBone 0301-620X/91/l059 $2.00 JBoneJoint Surg[Br] 1991 ; 73-B:162-8.
162
of Bologna
and
Joint
Surgery
2. Plain radiograph sarcoma shows a roundish mass with some stippled the deltoid. Case
THE
JOURNAL
ofa myxoid chondroslightly radiopaque calcification within
OF BONE
for
the radiographs a soft-tissue mass
mesenchymal.
R. Casadei, MD, Fellow First Orthopaedic Clinic M. Ricci, MD, Fellow Bone Tumour Centre P. Ruggieri, MD, Orthopaedic Surgeon First Orthopaedic Clinic R. Biagini, MD, Orthopaedic Surgeon First Orthopaedic Clinic S. Benassi, ScD, Biologist Bone Tumour Centre P. Picci, MD, Oncologist Bone Tumour Centre M. Campanacci, MD, Director First Orthopaedic Clinic of the University Bone Tumour Centre Istituto Ortopedico Rizzoli, Bone Tumour 1/10, Bologna, 40136, Italy.
I
for the seven women and two men aged 32 to 62 years. Eight tumours were in a lower limb and only one in an upper limb. Pain and tenderness were the main symp-
AND
JOINT
SURGERY
CHONDROSARCOMA Table
I. Clinical
Age(yr) and sex
Case Myxoid 1
details
of nine
patients
Site
chondrosarcoma 62 F
IA
Vastus lateralis Triceps
IA
32 F
3
45
4
48
F
5
46
M
Mesenchyma/ 6
chondrosarcoma 28 F Leg
F
with
Stage
Vastus medialis Deltoid
2
OF THE SOFT soft-tissue
Wide excision Wide excision Wide excision Marginal
lB
surae
163
chondrosarcoma Time to metastasis (mth)
Treatment
IA
TISSUES
Follow-up (mdi)
-
19
-
36
-
60
-
108
-
132
excision*
Vastus medialis
IA
Marginal excisionS
IIB
Marginal
Result
Tumour free Tumour free Tumour free Tumour freet Tumour freet
21
33
Died
0
13
Has metastases
6
48
Died
15
87
Died
excision*
7
37 F
Buttock
IIIB
8
55
Leg
IIB
9
27 F
Buttock
IIB
Hindquarter amputation (wide)
* local
M
:see text
recurrence
t free of tumour
Marginal excision Chemotherapy Marginal excision
for details
after excision
oflocal
with no distinctive features (cases 1, 4 and patient (case 2) minute stippled calcifications
recurrence
5). In one were seen
CT scans were performed in six cases, showing a soft-tissue mass in the patient with normal radiographs and contributing to pre-operative staging by demonstrating different densities of tumour and surrounding soft
(Fig. 1). In mesenchyma!
chondrosarcoma
radiographic finding irregular calcifications the lesion
was
cobweb-like
the
most
ill-defined, calcification
with
blurred
(Fig.
3).
margins
Fig.
Case
73-B,
No.
1, JANUARY
1991
and
thin,
7
tissue and the relationship bundles. Usually the tumour but in two with bone,
patients without
showcalcifica-
to bone and was distant
(cases 6 and any erosion
Fig.
2
6. Mesenchymal chondrosarcoma ing a large ovoid mass with irregular tion between tibia and fibula.
VOL.
frequent
was an ovoid mass, with numerous (cases 6, 8 and 9, Fig. 2). In case
7) the mass was in contact or penetration (Fig. 4).
3
Case 7. Mesenchymal chondrosarcoma buttock showing net-like calcification a mass with ill-defined margins.
neurovascu!ar from the bone
of the within
R. CASADEI,
164
Bone-seeking
isotope
M. RICCI,
scans,
P. RUGGIERI.
available
showed uptake in all patients. activity within the lesions showing two cases where tumour extended
in seven
R. BIAGINI,
S. BENASSI,
P. P1CC!,
M. CAMPANACCI
cases,
There was increased calcification. In the to the periosteum,
isotope scans also showed a reactive uptake in the adjacent bone. Gross findings. The five cases of myxoid chondrosarcoma presented as ovoid, soft, nodular masses, well circumscribed by a distinct pseudocapsule. On section the surface appeared translucent, gelatinous, and grey to brown in colour with large haemorrhagic and sometimes necrotic areas. The size of the tumours ranged from 5 to 8cm. The mesenchymal chondrosarcomas, were multilobulated and circumscribed, firm in consistency with no well-defined pseudocapsule. Cut sections of specimens showed
a
pale
cartilaginous necrosis were
grey,
fleshy
surface
with
foci, which creaked on cutting. abundant while haemorrhagic
Fig.
scattered
4
CT scan of Case 6 (Fig. 2) shows a partially mass in contact with the bone without penetrating
Areas of zones were
sometimes eosinophi!ic In case
tive tissue capsule, which had resulted from the expansile growth of the tumour. Fibrous septae of variable thickness with a few small blood vessels extended in from
scanty myxoid ground substance. Anaplasia and pleomorphism were rare and mitotic figures were absent. We observed no differentiated chondrocytes within distinct lacunae. Mucoid material was abundant in three patients
narrow vacuolated chromatic
rim
round of
by variable was weakly
deeply
cytoplasm nucleus.
fine anastomosing tumour a lace-like
Within the or slightly
eosinophilic around
Typically strands appearance.
amounts basophilic
lobules, elongated. the
the and
the and
small, cells
cells There
and in two, the typical texture ofmyxoid was disturbed by large haemorrhagic
sometimes ovoid,
were
were was a
arranged
and occasional haphazardly argyrophilic
hyperin
small nests giving the These cells were separated
of mucoid ground with haematoxy!in
stain was of glycogen
substance which and eosin, but
chondrosarcoma
#{149}1
a
probably
homogeneous or whorls
with
chondrosarcoma and necrotic areas
calcification. Silver stains arranged reticulin fibres and material within the myxoid
always positive, in the cytoplasm.
Mesenchymal
of
it.
scanty. The tumour size ranged from 10 to 20 cm. Histological findings. Myxoid chondrosarcomas were multilobular and well circumscribed by a condensed connec-
the pseudocapsu!e. small, uniform,
contained small droplets material (Fig. 5). 2 there were cells in clusters
calcified
due showed
showed a few some granular matrix. PAS to the
presence
a characteris-
Case 2. Myxoid chondrosarcoma (Fig. 1) showing the characteristic alignment of tumour cells in strands and cords separated by large amounts of myxoid material. The vacuoles within the myxoid ground substance are extracellular and should not be confused with lipoblasts. Note the absence ofvessels (haematoxylin and eosin, x 200).
a
T*
Fig. 5
THE
JOURNAL
OF BONE
AND
JOINT
SURGERY
CHONDROSARCOMA
tic pattern oftwo basic round or spindle-shaped differentiated mesenchyma! of
cellular cells
tumour component
elements : undifferentiated and small nodules of well-
cartilage. showed
haemangiopericytoma
OF THE
(Fig.
7 showed
plasm. figures
nuclei
In all cases and dilated
Cartilaginous abundant
and
scanty,
poorly
outlined
there were numerous vascular clefts. foci
in only
were
two.
present
These
in
nodules
a
mitotic
a!!
tumours,
were
well
there were negative.
pattern
of
large Silver
reticu!in
undifferentiated
necrotic stains fibres
cells
and
zones. showed around
but defined
groups
of
vessels.
cases
of myxoid
chondrosarcoma
were
treated
by
wide excision. Al! these patients are alive and free of tumour after an average of 38 months (19 to 60). In case 4 a marginal excision had been performed elsewhere and at
12 months
a local
required amputation. free at nine years. The fifth patient with the diagnosis developed
a local
recurrence This
in the
patient
had a marginal of ‘myxoma’ recurrence
;
biopsy
popliteal
is alive excision and five then
and
fossa disease-
elsewhere, years later provided
the
again excised our care with
free
of tumour
No. 1, JANUARY
1991
marginally. a further
Of the four one
was
treated
surgical region, anterior there
A marginal therapy.
at 1 1 years from
margin. attached
the
a wide
In 1986 large local
the patient recurrence
vessels. A wide bypass. The
excision surgical
from
excision was performed, The patient is alive and the
first
operation
and
at
last.
cases of mesenchymal chondrosarcoma, by hindquarter amputation with a wide This patient to the iliac
portion of the thigh. was a single pulmonary
had a mass wing and At
in the g!utea! involving the
the time metastasis.
of diagnosis, This was
excised. Ten months later multiple pulmonary metastases developed ; the patient was treated with chemotherapy and radiotherapy. He is still alive at 1 3 months.
margins in two
other
three had
patients conservative
with mesenchyma! surgery. The
were ‘marginal’ and a local cases at three and 12 months
recurrence respectively.
chonsurgical developed In case
6 radiation
therapy was used in an unsuccessful attempt to control the local recurrence ; finally an amputation was performed. In case 9 the recurrence was adherent to the periosteum of the iliac wing without involvement of the bone : wide excision with a partial resection of the iliac wing
was
developed involvement
performed.
In
these
in lung, liver and of supraclavicu!ar
Fig.
73-B,
and
However, the excision was in 1984 a further recurrence
the femora! an arterial
Case 6. Mesenchymal chondrosarcoma (Fig. 2) showing, on the left, areas of welldifferentiated cartilage surrounded by undifferentiated cells in a highly cellular arrangement ; on the right, an alveolar and haemangiopericytoma-like pattern (haematoxylin and eosin, x 50).
VOL.
chondrosarcoma
margins of this specimen were confirmed to be wide, but three years later a further small recurrence developed
The drosarcoma
TREATMENT Three
of myxoid recommended. marginal and
in the thigh involving was attempted with
six months
PAS stain was a well-defined small
was than
close to the bypass. followed by radiation
in two cases while in another two they had ill-defined margins that blended with the mesenchymal areas. In three cases they followed lobules of!ow-grade malignancy (grade 1 to 2). Haemorrhagic areas were rare but in three cases always
165
diagnosis
developed, came under
cyto-
atypical
TISSUES
excision no more
many
areas ofdense cellularity reminiscent ofEwing’s sarcoma, and in case 6 there were round cells arranged like lymphoma. Mesenchyma! cells had ovoid or elongated hyperchromatic
correct
In cases 8 and 9, the the histological pattern 6). Case
SOFT
6
three bone
and lymph
cases
metastases
case 6 also had nodes. These
166
R. CASADEI,
patients
died
at
Chemotherapy when metastases
33,
has been appeared
M. RICCI,
48,
87
P. RUGGIERI,
months
used only in one in bone.
R. BIAGINI,
from
surgery.
patient
(case
Enneking 8)
is a quite a pre-existing
rare tumour chondroma
of the soft tissues (Enzinger and Weiss 1983). Recently Enzinger and Shiraki (1972), Hajdu (1979) and Campanacci, Bertoni and Bacchini (1981) have distinguished two typical sub-groups. Clinical findings. Myxoid chondrosarcoma occurs more frequently in lower limbs, deep to the fascia in muscular compartments ger and Weiss
(stage 1983).
A) (Campanacci Mesenchymal
arise in any mesenchymal Hajdu 1979), sometimes in and Garret 1963). When it usually arises in the soft tissues (stage B, Enneking
tissue (Guccion et a! 1973; an irradiated area (Brenner is found in the extremities, it areo!ar extracompartmental 1983). Myxoid chondrosar-
coma occurs more frequently (Uehara and Becker 1960; Angerva!l, Enerb#{228}ck and Ferenczy
1978
et a! 1981 ; Enzinchondrosarcoma may
in adult Enzinger Knutson
; Campanacci
and elderly males and Shiraki 1972; 1973 ; Mehio and
et a! 198 1 ; Amir
et a! 1985)
although in our series we observed a 4 to 1 female to male ratio. The mesenchyma! variety occurs more often in young adult females (Salvador, Beabout and Dahlin 1971 ; Guccion et a! 1973; Campanacci et a! 1981; Enzinger Pain
and and
Weiss 1983; Nakashima tenderness are the most
observed
in both
types
et a! 1986). frequent symptoms
of extraskeletal
chondrosarcoma
(Salvador et al 1971 ; Campanacci et a! 1981 ; Enzinger and Weiss 1983; Amir et a! 1985). In our patients the average duration of complaints was 1 7 months in myxoid and nine months in mesenchymal chondrosarcoma. Imaging. A well-defined soft-tissue neoplasm, often speckled with calcification, is more indicative of a mesenchyma! than a myxoid chondrosarcoma because in this second type there is no intratumora! calcification or only and
minor Shiraki
stippling 1972;
(Uehara Campanacci
1985). In mesenchymal floccules are different pattern typical (Campanacci
Becker 1960 ; Enzinger et a! 1981 ; Amir et a!
chondrosarcoma from the granular
the flecks and or ‘popcorn’
of the osseous type of chondrosarcoma et a! 1981 ; Bertoni et a! 1983; Enneking
1983). In myxoid chondrosarcoma radiographic features does diagnosis et a! 198!
and
from other Enzinger
;
not
soft-tissues and Weiss
the lack of distinctive allow a differential
sarcomas 1983).
(Campanacci
Intrinsic radiopacities may suggest lesions which contain amorphous calcification, either benign tumours, such as angioma, synovial chondromatosis, or chondroma (Goldenberg
et
al
1967;
Wi!ner
1982),
or
as
tumoral
ca!cinosis or soft-tissue calcification in secondary hyperparathyroidism (Bertoni et a! 1983), or malignant as in extraskeletal fibrosarcoma (Goldenberg et a! 1967;
P. PICCI,
1983),
M. CAMPANACCI
extraskeleta!
al 1983), or synovial Enzinger and Shiraki 1983; Enneking 1983; Isotope
DISCUSSION Extraskeleta! chondrosarcoma arising ‘de novo’, and not from
S. BENASSI,
scans
osteosarcoma
(Bertoni
et
sarcoma (Go!denberg et a! 1967; 1972; Wilner 1982; Bertoni et a! Nakashima et a! 1986).
show
uptake
in both
types
of tumour
but is more pronounced coma, sometimes with
in mesenchymal increased activity
chondrosarin the adjacent
bone because the tumour In myxoid chondrosarcoma
was extended angiography
to the periosteum. delineates the
mass
and
the reaction
in the surrounding
soft
tissues
and
shows the relationship neurovascular bundles. strongly because the comparison with its (Enneking 1983). In ma! chondrosarcoma
of the tumour with the adjacent The tumour itself does not inject interior of the mass is avascular in peripheral reactive vascularisation contrast, angiography in mesenchyshows a hypervascu!arisation of the
neoplasm
broadened
with
many
capillaries
(Campanacci
eta!
1981). CT scans are necessary for surgical planning demonstrate differences in density in comparison the adjacent soft tissues and with other soft sarcomas (Enneking very low fluid density whereas density
they with tissue ;
1983). The myxoid variety has due to the richly mucinous material,
mesenchymal due to calcification.
chondrosarcoma Only in a few
shows reported
a
a high cases
is there evidence of erosion of the adjacent bone (Stout and Verner 1953; Go!denberg et a! 1967; Steiner, Mirra and Bullough 1973; Amir et a! 1985). Differential diagnosis. Myxoid chondrosarcoma may be mistaken
for
other
myxoid liposarcoma, liposarcoma has
richly
mucinous
myxoma a plexiform
tumours
such
and chordoma. vascular pattern,
as
Myxoid many
lipoblasts and no stainable mucin after previous treatment of the sections with hya!uronidase (Campanacci et al 1981). Histologically, chordoma is very similar to myxoid chondrosarcoma because it also contains mucoid material with the same staining characteristics. However, the usual site (skull, spine and sacrum) and the presence of physaliferous cells, are useful for the differential diagnosis (Mehio and Ferenczy 1978). A myxoma can also be confused with a myxoid chondrosarcoma, but it has more scattered and less defined cells not arranged in cords, and the mucoid material has the same staining characteristics Shiraki
as
1972; Hajdu The differential
sarcoma sarcoma,
myxoid
!iposarcoma
is made from haemangiopericytoma, !ymphoma, rhabdomyosarcoma,
coma, and high-grade liposarcoma. drosarcoma may often be haemangiopericytoma because of pattern (Goldman 1967; Salvador et a! 1973; Campanacci et a! 1981 ; Enzinger
(Enzinger
and
Weiss
1983;
differs in its well-defined poor intercellular reticulum. (Steiner et a! 1973 ; Fu THE
and
1979; Campanacci et a! 1981). diagnosis of mesenchymal chondro-
Nakashima cartilaginous Electron and Kay
JOURNAL
OF BONE
Ewing’s synovial sar-
Mesenchymal chonmistaken for a its similar cellular a! 1971 ; Guccion et Bertoni et a! 1983; et a! 1986), islands microscope 1974) show AND
JOINT
but
it
and the studies that in SURGERY
CHONDROSARCOMA Table tissues
II.
Differences
between
OF THE the two
SOFT
sub-groups
Small
mass
Large
Radiograph
Little
or no calcification
Irregular
CT scan
Low homogenous
Bone
Faint
Histology
Strands and cords round cells within amount of mucoid
Local
Radiotherapy
mesenchymal cells and islets of well-differentiated cartilage with central calcification Quick invasive growth
Probably
Useless
Metastases
Rare
and
Prognosis
Good
vascular
cells have the
hypervascularity
Radical excision amputation
after
the
density uptake
Undifferentiated
of large matrix
growth
Rare
undifferentiated ; they do not
calcification
Diffuse
Wide
recurrence
chondrosarcoma
the result of vascular spaces
High
Hypervascular margin Avascular centre
mass
Irregular
uptake
Slow
mesenchymal
density
Angiography
Chemo/
soft
35
scan
167
ofchondrosarcoma
Myxoid
(yr)
Age
TISSUES
wide
excision
small
or
useful
Frequent
late
Common
and
early
Bad
features
are
growing around characteristics of
often be treated by limb salvage surgery. of extraskeleta! chondrosarcoma often putation (Salvador et a! 1971 ; Enneking
Local recurrence requires an am1983).
pericytes. Other malignant round cell tumours lack the foci of cartilaginous differentiation (Salvador et a! 1971; Steiner et a! 1973 ; Bertoni et a! 1983 ; Nakashima et a! 1986). An alveolar and haemangiopericytoma-like texture may also be observed in synovial sarcoma, but there
There are no definitive data about the role of chemotherapy or radiotherapy in addition to an adequate surgical excision. Campanacci et al (1981), Bertoni et a! (1983), and Enzinger and Weiss (1983) suggest the use of
is often appearance
In myxoid chondrosarcoma the rate of metastasis is lower (20%, Enzinger and Shiraki 1972) than in the mesenchymal variety (50%, Salvador et al 1971). We found a more striking difference : no metastasis compared
a
biphasic pattern, and proliferating
with a pseudoglandular spindle-shaped cells (Gold-
man 1967 ; Guccion et a! 1973 ; Enzinger and Bertoni et a! 1983 ; Nakashima et a! 1986). Treatment and prognosis. Staging studies omitted lesional
and tumours are excision (Salvador
treated by marginal et a! 1971 ; Smith
adjuvant
therapy
Weiss
1983;
are
often
with
100%
or intraet a! 1976;
we!!
localised,
Enneking 1983 ; Amir et a! 1985). The reported rate of local recurrence and metastasis, therefore, varies from 20% to 60% in soft-tissue chondrosarcomas (Stout and Verner 1953 ; Kauffman and Stout 1963 ; Goldenberg et
in mesenchymal
respectively. the
When patient
may
chondrosarcoma.
a metastasis be treated
is unique
and
by pulmonary
lobectomy or local excision, because of the slow growth ofthe tumour cells (Wu et a! 1980). The patients must be followed up for a long time to evaluate correctly the survival and recovery, because coma may develop a recurrence after surgical al 1981).
percentage
coma of bone is equally aggressive (31% of recurrences) as myxoid chondrosarcoma of soft tissue (40% of recurrences) but has a worse prognosis (39% of metastases as against no metastases). According to Enzinger and
coma series)
of recurrence
either
(30%, Enzinger and or in the mesenchymal
a! 1971 ; 40%, Wide surgical myxoid sub-type acci et al 1981),
Guccion resection
in myxoid
chondrosar-
Shiraki 1972 ; 100%, in our variety (33%, Salvador et
et al 1973 ; 66%, in our is the treatment of choice
(Enzinger whereas
series). for the
and Shiraki 1972 ; Campanfor the mesenchymal variety
radical excision is preferable (Salvador et a! 1971; Campanacci et a! 1981 ; Bertoni et a! 1983 ; Enneking 1983 ; Enzinger and Weiss 1983 ; Nakashima et al 1986), because the tumour is frequently extracompartmenta!. For this reason, mesenchymal chondrosarcomas cannot VOL.
73-B, No.
1, JANUARY
1991
a
From
(Wu
et a! 1980;
chondrosarmany years
a! 1967 ; Guccion et a! 1973 ; Wu, Collon and Guise 1980). In our series it occurred in 44% and was closely related to tissue margins at the first surgical treatment. A marginal or intralesiona! excision has a high
tumour
treatment
soft-tissue or metastasis
a study of all chondrosarcomas centre (Mercuri et a! 1988) central
Campanacci
et
treated in our chondrosar-
Shiraki (1972), Smith et a! (1976), and Amir et al (1985), mesenchymal chondrosarcoma has a higher malignancy when it is localised in soft tissues : 14% recurrence and 7 1% metastasis in mesenchyma! chondrosarcoma of bone as against 50% and 100% in the soft-tissue type. In our series, mesenchymal chondrosarcoma has a worse prognosis than both the myxoid variety and osseous chondrosarcomas.
168
R. CASADEI,
M. RICCI,
Chondrosarcoma logically well defined
of soft tissues and distinct
chondrosarcoma (Table II). They
mesenchymal in both clinical
features
and
and differ
P. RUGGIERI,
includes sub-types,
two
R. BIAGINI,
histomyxoid
chondrosarcoma and radiographic
in prognosis.
special project ‘Oncology’ was supported by grant number 124.44 from the Italian National Council of Research. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article.
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