aggressive scapular chondroblastoma with secondary metastasis

EUROPEAN JOURNAL OF MEDICAL RESEARCH 1

AbstractChondroblastoma is a benign bone tumor, accountingfor approximately one percent of all benign bone tu-mors. It mostly occurs in typical locations such as longbones. Malignant transformation including metastasishas been described in only a few cases. Therefore, wereport a unique case of chondroblastoma with tumormanifestation in the 7th decade of life, location of thetumor in the scapula and occurrence of metastasis inthe soft tissue of the mandible branch. Due to aggra-vation of the clinical course, a scapula en bloc resec-tion was performed. The differential diagnosis is dis-cussed and the current literature concerning malignanttransformation of chondroblastoma is reviewed.

Key words: Chondroblastoma, malignant transforma-tion, en bloc resection therapy, metastasis

INTRODUCTION

Chondroblastoma is a benign bone tissue tumor,which was first reported by Codman in 1931 in termsof an “epiphyseal chondromatous giant cell tumor”.This rare tumor entity accounts for approximately onepercent of all benign bone tumors, most frequentlyfound in young male patients with a peak incidence inthe second decade of life. Although chromosomal ab-normalities have been reported, no specific alterationhas been detected so far. Typical tumor locations arethe long bones, especially femur, tibia and proximalhumerus. Common clinical symptoms are pain and adecreasing range of motion of adjacent joints. In con-ventional radiographs, signs of asymmetric epiphysealdestruction, often including bone expansion and scle-rotic margins, can be seen. In histological staining, thepredominant cells are most similar to epiphyseal chon-drocytes, expressing distinct morphological featuresfor differentiation. Although most authors assume thetumor being of essentially benign character, 11 casesof malignant transformation including metastasis havebeen reported in the literature (Table 1). In this con-text, the present case appears unique due to 1) thescapular location of the tumor, 2) the occurrence ofmetastasis in the soft tissue of the mandible branch,and 3) a course of disease lasting more than 40 yearswith tumor manifestation in the 7th decade.

CASE REPORT

A 68-year-old man in good physical condition present-ed with limitation of left shoulder mobility. Physicalexamination revealed a tumor along the dorsolateraledge of the left scapula with atrophy of the left shoul-der girdle musculature compared to the right side. Thepatient felt pain in palpation of the lesion and at thelateral shoulder edge. Active adduction/abduction ac-counted for only 20–0–30, the passive one for20–0–50 degrees with a restriction of rotational move-ment. The neurovascular status was inconspicuous ex-cept a significant strength loss of the left versus theright hand. No lymphadenopathy, fever, chills, orweight changes were present; the laboratory findingswere within normal ranges.

Computed tomography (CT) showed a huge tumormainly of soft tissue appearance with significant ossifi-cation and partial destruction of the left scapula. In a99mTc bone scintigraphy, a significant enhancementof activity in the tumor surrounding in terms of theproximal left scapula and a large expansion into thesuprascapular tissue was detected. In magnetic reso-nance imaging (MRI), a lesion of 11 x 7 x 9 cm in size,partly lobulated and nodular with expansion startingfrom the spina scapulae, was visualized with distinctareas in T1- and T2-weighted images before and afterapplying contrast medium indicating necrotic areas.The tumor itself showed a distinct contrast enhance-ment with infiltration of the rotator cuff and the originof the subscapularis muscle. The branches of brachialplexus as well as the subclavian artery appeared with-out infiltration. The histological examination of abiopsy established the diagnosis of Langerhans' cellhistiocytosis. After 15 cycles of fractional radiation upto a maximum of 30 Gy, restaging showed a neitherclinical nor radiological regression of the tumor 6months after primary diagnosis. A second biopsy didnot confirm the diagnosis of a Langerhans' cell histio-cytosis, but diagnosed a benign chondroblastoma.Since the patient refused to undergo surgery, he wasreassigned every 6 months for control examinations.Three years after the patient's first presentation, a sig-nificant clinical aggravation of the tumor was ob-served (Fig. 1). For the mobility of the shoulder, activeadduction/abduction now accounted for 20–0–10, the

Eur J Med Res (2006) 11: 1-7 © I. Holzapfel Publishers 2006

AGGRESSIVE SCAPULAR CHONDROBLASTOMA WITH SECONDARY

METASTASIS – A CASE REPORT AND REVIEW OF LITERATURE

C. Kirchhoff1, S. Buhmann2, T. Mussack3, J. Müller-Höcker4, M. Schmitt-Sody1, V. Jansson1, H.-R. Dürr1

1Orthopädische Klinik und Poliklinik, 2Institut für Klinische Radiologie, 3Chirurgische Klinik und Poliklinik-Innenstadt, 4Institut für Pathologie,

Klinikum der Ludwig-Maximilians Universität München, Munich, Germany

EUROPEAN JOURNAL OF MEDICAL RESEARCH2

Tab

le 1

.Tab

le o

f all

patie

nts

with

his

tolo

gica

lly p

rove

n m

etas

tase

s of

a c

hond

robl

asto

ma,

rep

orte

d in

the

liter

atur

e si

nce

1931

Au

thor

(R

ef.)

Ag

e,

Site

of

Pri

mar

yC

ours

e an

d T

reat

men

tIn

terv

al t

o F

ollo

w U

pSe

xm

etas

tase

s

Dah

lin D

.C. [

5]-

Pelv

is1)

Rep

eate

d co

urse

s of

rad

iatio

n th

erap

y; 2

) Mul

tiple

pul

mon

ary

met

asta

ses

58 m

onth

sD

eath

due

to p

ulm

onar

y m

etas

tase

sG

awlik

Z. [

8]6

fem

Prox

. Hum

erus

1) I

nter

scap

ulot

hora

cic

ampu

tatio

n; 2

) Med

iast

inal

and

pul

mon

ary

met

asta

ses;

3

mon

ths

Dea

th d

ue to

wid

espr

ead

pulm

onar

y3)

Tho

raco

tom

ym

etas

tase

sG

reen

P. [

9]13

fem

Prox

. fem

ur1)

Loc

al r

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) Tho

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mon

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Stab

le p

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sion

sH

uvos

A.G

. [10

]16

fem

Dis

t. fe

mur

1) C

uret

tage

; 2) L

ocal

rec

urre

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with

pul

mon

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nodu

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mon

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No

furt

her

met

asta

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3) C

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; 4) M

ultip

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dule

s5)

Tho

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mbh

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NA

. [13

]27

fem

Tal

us, p

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y 1)

Exc

isio

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talu

s; 2

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rs. L

ater

thor

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3 yr

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pul

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ecur

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pul

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nodu

leJo

shi D

.D. [

15]

17 fe

mFi

bula

1) R

esec

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of p

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B, t

hora

cosc

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res

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tabl

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esec

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of b

one

and

soft

tiss

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tase

sm

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L.B

. [16

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mal

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uret

tage

; 2) L

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rec

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and

pulm

onar

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th d

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met

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sis

15 y

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3) T

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tim

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tage

and

res

ectio

n of

the

prim

ary

afte

r 1s

tcu

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age

Kha

lili K

. [17

]43

mal

eR

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) Diff

use

soft

tiss

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s (s

calp

, nec

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tock

)5

yrs.

No

loca

l nor

pul

mon

ary

met

asta

ses

Kun

ze E

. [18

]33

mal

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lvis

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mon

ary

met

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12 y

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Stab

le p

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y le

sion

sK

yria

kos

M. [

19]

9 m

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ia1)

Cur

etta

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one

pack

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Tho

raco

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ic r

esec

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of tw

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2) L

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3) S

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mot

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wed

by

cure

ttag

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the

tibia

l rec

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Ram

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A. [

22]

29 m

ale

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) Mul

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3) R

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and

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blat

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Rid

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R.J.

[23]

14 fe

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Scha

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4]32

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Swee

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26]

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29]

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0 yr

s. L

ater

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5) T

hora

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my

Wel

lman

n K

. [30

] *29

mal

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] *38

mal

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t: D

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ann

[30]

and

Wirm

an [3

1]: T

he o

nly

two

auth

ors

repo

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g of

an

chon

drob

last

oma

of th

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apul

a w

ith m

etas

tase

s) a

nd o

f th

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tient

s’cl

inic

al fe

atur

es.

passive one for 20–0–20. A weight loss of 2kg during8 weeks was registered. Additionally, a new 2x2cmsized soft tissue tumor of the right mandible branch,which was first regarded as atheroma or lesion of thesalvatory gland, was found. CT showed a maximumprimary tumor size of 11 x 5 x 10cm with the main fo-cus at the dorsocranial scapula and massive destruc-tion of the medial scapular third. MRI revealed a pro-gressive and partly neoplastic chondroblastoma with astrong contrast enhancement at the margins (Fig. 2).Another fine needle biopsy proved no alteration ofhistology. Suspecting a malign transformation to achondrosarcoma, an en bloc scapulectomia involvingresection of surrounding soft tissue was performed,The resected specimen had 1255g in weight and22 x 17 x 9cm in size. Histological examination showeda population of monomorphic medium-sized cellswith slightly recessed nuclei, broad and eosinophiliccytoplasm, and sharply defined cell boarders. Theywere presenting mainly giant cells of the osteoclastictype without atypia, but widespread necrosis and con-sistent mitosis (mitotic rate 10/10 HPF). Birefringentcrystal material compatible with calcium pyrophos-phate focally accumulated. Moreover, areas with pro-duction of chondroid matrix and enclosure of differ-entiated chondroid cells were recognized (Fig. 3).Bone destruction, infiltration of adjacent muscles andsubcutaneous soft tissue were found thus establishingthe diagnosis of an aggressive chondroblastoma. Thetumor above the right mandible branch showed slightCT enhancement. Histology revealed the identicalmorphology of the scapular chondroblastoma.

Postoperatively, neither lung metastasis nor any oth-er filia were seen in CT. However, the right mandibletumor showed slight enhancement in 99mTc bonescintigraphy with the histological diagnosis of a metas-tasis of the scapular chondroblastoma. Two and a half

years after the final surgery, the patient still presentedfree of metastases with an acceptable range of motionregarding his left upper limb.

DISCUSSION

The first description of chondroblastoma was given byCodman in 1931, who designated it as “epiphysealchondromatous giant cell tumor” [3]. Jaffe and Licht-enstein differentiated in 1942 the chondroblastomafrom giant cell tumors and established the term “be-nign chondroblastoma” [12]. Chondroblastoma is arare tumor with an incidence of 1% of all benign bonetumors. In up to 95% it occurs in the second decadeof life with a male:female ratio of 2 : 1 [24]. Especiallyin older patients, there were reported a few atypicalcases of chondroblastoma, which tend to involve un-usual sites and have a greater tendency to expand theaffected bone. This observation was confirmed by thepresent case in an 68-year old male. Chondroblastomahas a predilection for the epiphysis of the femur,humerus, tibia and tarsal bones. Only 16% of cases arefound in flat bones [1, 5]. Scapular location of a chon-droblastoma, as described in our case, has been re-ported since 1931 in only eleven cases (Table 1). Here-by, our patient is the oldest reported case of scapularaffection in the entire described 12 cases at all. Mostpatients reported pain and local tenderness associatedwith limitation of motion of the adjacent joints. Apathologic fracture as the primary presentation is ex-ceedingly rare, and local swelling is present in less than10 percent [11].

Histogenesis of chondroblastoma is controversial,but derivation from epiphyseal cartilage cells ratherthan from reticulo-histiocytic cells seems to be most


Fig. 1. Photography of the patient’s back before surgery. Atthe upper margin of the left scapula a tumor mass is notable(indicated by the red arrow). The affected shoulder appears tobe elevated in level compared to the opposite, right shoulder.Underneath the tumor mass, a mark of approximately 5cmlength is visible, most likely due to an ancient surgical proce-dure. A defect with consecutive loss of mass of the deltoidmuscle can be seen on the left compared to the right deltoidarea.

Fig. 2. Radiograph of the left shoulder showing a huge calci-fied tumor mass above the clavicle. The tumor shows a scle-rotic inferior margin and extension into the soft tissue, aswell as into the glenoid fossa

likely [5, 24]. The histological diagnosis of typicalchondroblastoma is usually not difficult due to theircharacteristic appearance with rounded or polygonalchondroblasts, multinucleated giant cells, andeosinophilic chondroid extracellular matrix with focal

(chicken wire) calcification [4]. Nonetheless, it is im-portant to be aware of a wide morphologic spectrumcausing possible diagnostic problems. Therefore, thedifferentiation between chondroblastoma and giantcell tumor can be difficult in case of the former con-


Fig. 3. Magnetic Resonance Images (MRI) of the left scapula in the parasagittal plane: On the left side (3a, b), MR-images of theyear 2000 are shown: A huge tumorous mass, in the soft tissue beneath the deltoid muscle is visible. The lesion measures11x7x9 cm in size. On fat-suppressed images (3a), a hyperintense, inhomogeneous signal results whereas after contrast applica-tion (3b) a rich enhancement is shown indicating a necrotic central zone. On the right side (3c, d), MR-images 3 years later aredisplayed: A second tumor nodule is now recognizable, located just dorso-caudal to the primary tumor mass (indicated by thered arrow). This nodule is of homogeneous hyperintense signal on fat-suppressed images (3c) and shows homogeneous contrastenhancement (3d) indicating viability.

a c

b d

taining a large number of giant cells and scanty chon-droid matrix. The epiphyseal location and lytic radi-ographic appearance may add to the confusion, butchondroblastoma usually occur in patients youngerthan 20 years with open epiphyseal plates, often in-volving less than one half of the epiphyseal diameter.The mononuclear cells in chondroblastoma will con-tain irregular, ridged nuclei with longitudinal clefts,while in giant cell tumors the cells resemble histiocyteswith nuclei, similar to those of the giant cells [2]. Fo-cally, a network of calcification (chicken wire) betweenthe tumor cells exist, whereas clumps of calcificationwere observable in some areas [22].

Chondroid plaques are not a feature of giant cell tu-mors. In difficult cases, the demonstration of S100positivity in chondroblasts will be helpful [28]. One ofthe most difficult entities being differentiated from achondroblastoma is the Langerhans' cell histiocytosis,which was considered the primary diagnosis in our pa-tient and consecutively led to inadequate initial treat-ment [7]. Cytologically, chondroblasts exhibit roundedcontours and folded or reniform-shaped nuclei, evencontaining prominent grooves. Furthermore, binucle-ate and multinucleate forms are usually present. Thesecytomorphologic features also characterize the diag-nostic histiocytes of Langerhans' cell histiocytosis,which is not associated with any form of matrix pro-

duction [7]. As shown in the present case, immunohis-tochemical results may even not be conclusive for theexclusion of Langerhans' cell histiocytosis showing un-equivocal expression of CD1a.

In an older group of age, the differential diagnosisof a low graded chondrosarcoma may also be consid-ered. However, concerning cytology the cells in achondrosarcoma are larger, with mild to moderatepleomorphism and present in lacunae. Binucleate andmultinucleate cells are frequent along with the pres-ence of hyaline cartilage [20].

Features supportive for a benign lesion will be theabsence of abnormal mitoses and a low mitotic count.The mitotic count of chondroblastoma does not ex-ceed 5/10 HPF in 81% of tumors. In cases with un-usual histologic features, correlation with radiographicfeatures will usually help with the diagnosis. Radiologi-cally, chondroblastoma are usually presented as well-defined eccentric lesions in epiphyseal location with arim of sclerosis with or without punctuate calcifica-tions occurring in approximately 60%. Occasionally,large bubbly bone expansion with varying amounts ofstippled calcifications and periosteal reaction can beevident on radiographs and a secondary aneurismalbone cyst should be considered [5, 6]. A distinctivefeature of chondroblastoma on MRI scans is a pre-dominant low-to-intermediate signal intensity on T1-


Fig. 4. Histological HE staining: Numerous chondroblastswith eosinophilic cytoplasm and distinct cell borders as wellas mitotic activity are shown. Giant cells of osteoclastic type,varying in size are also visible (4a). S 100 Immunohisto-chemistry: Cytoplasmic staining in the mononuclear cells isshown (4b). CD 68 Immunohistochemistry: The mononu-clear cells remain unstained. The staining is confined to theosteoclastic giant cells (4c).

a c

b

weighted images with heterogeneous intermediate sig-nal on T2-weighted images, with only scattered foci ofhigh signal intensity. This low-to-intermediate signalintensity appears due to cellular chondroid matrix andalso calcification [14].

According to malignant tumor behavior concerningthe spread of metastases, there are a few documentedfatal cases of chondroblastoma showing pulmonarymetastasis of the same histologic characteristics as theremoved tumor, [10, 13, 19] thus presenting a metasta-tic chondroblastoma. Those cases have in commonthat surgical therapy always happened prior to the ap-pearance of metastases. Therefore, metastases mayrepresent a vascular transport phenomenon duringvigorous curettage [13]. Our patient also seems to beone-of-a-kind due to the fact that except fine needlepuncture no tumor manipulation was performed.Chondroblastoma is generally treated by curettagewith or without bone grafting with a curative result inapproximately 90% [19]. Cementation or excision andreconstruction may be required for large lesions. Therecurrence rates for chondroblastoma in previousstudies ranged from 5.7% to 20%; in case of sec-ondary aneurismal bone cyst identification, an evenhigher local recurrence rate was reported [10]. In gen-eral, local recurrence is more likely to occur in flatthan in long bones. In some rare cases, chondroblas-toma grow quite large, like in our case, or tend to recurwith or without soft tissue seeding. These large and/orlocal recurrent lesions have been described as aggres-sive variants of chondroblastoma [10, 21]. One mightthink that in these cases a separate, own tumor entityis concerned whereas the cytopathological specificcharacteristics have yet not been described in literaturedue to its rare occurrence [21]. Also the present reportproves that in case of the chondroblastoma remaininguntreated, it can show distinct size-progression, andthus making an en bloc resection necessary, which mayresult in a loss of joint function and growth distur-bance due to the location in the epi- and metaphysealregion [22, 27].

SUMMARY

In summary, the need for a combined and extendedclinical, radiologic, and histologic approach to the cor-rect diagnosis of chondroblastoma is emphasized. Asshown in the present patient, most of the classic crite-ria such as typical location, age, histology and radio-logical appearance are not met in these cases. The pa-tient might be endangered for an inadequate primarytreatment leading to fatal consequences. Therefore, westrongly suggest multiple biopsies of the tumor to geta distinct histological diagnosis. However, the occur-rence of metastatic and malignant behaviour of chon-droblastoma is extremely rare. It should not be takenas an indication for radical therapy like en bloc resec-tion necessary for every primary appearance of chon-droblastoma.

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Received: December 7, 2005 / Accepted: February 6, 2006

Address for correspondence:Dr.med. Chlodwig KirchhoffOrthopädische Klinik und PoliklinikKlinikum GrosshadernLudwig-Maximilians Universität MünchenMarchioninistrasse 15D-81366 Munich, GermanyE-mail: [email protected]

Senior Author:Prof. Dr. med. Hans-Roland DürrOrthopädische Klinik und PoliklinikKlinikum GrosshadernLudwig-Maximilians Universität MünchenMarchioninistrasse 15D-81366 MunichE-Mail: [email protected]


aggressive scapular chondroblastoma with secondary metastasis

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