大 韓 放 용f 線 짧 學 會 註 第 26 卷 第 5 號 pp. 940 - 943 , 1990 Journal 01 Korean Rad iological Society, 26(5) 940 - 943 , 1990

Metachronous Osteosarcoma - A case report -

Myung Joon Kim, M.D. , Jin Suck Suh, M.D. , Chang Yun Park, M.D. , Department of Diagnostic Radiology, Yonsei University College of Medicine, Seoul, Korea

〈국문초록〉

후시성 골육종

-1 예 보고-

연세대학교 의 과대학 진단방사선과학교실

김 명 준·서 진 석·박 창 윤

후시 성 골육종은 매우 드문 질환으로 각 병변의 방사선학적 ， 조직 학적 소견은 일반적인 골육종과 차

이가 없다. 그러나 동시성 ， 다발성 골육종과는 이차 종양의 발생 시기 및 분포， 예후에 있어 차이 가 있

는 것으로 알려져 있다. 저 자들은 6세 여아에 발생 한 후시성 골육종 1예 를 경험하였기에 문헌 고찰과 함

께 보고하는 바이다.

- Abstract-

The metachronous osteosarcoma is a rare disease. The radiological and histological findings

of each lesion of metachronous osteosarcoma are not different from the usual osteosarcoma

But it is known to be different from the synchronous , multifocal osteosarcoma in regard to the

late development of secondary tumor, distribution of the tumors and prognosis. We report a

case of metachronous osteosarcoma in a 6 year old girl with review of the literatures.

Index Words: Bone neoplasms , diagnosis , 40.32

Extremities, osteosarcoma, 432.3221

Children, skeletal system

Osteosarcoma is a highly malignant bone

tumor , and it mainly involves around the knee

joint. Osteosarcomas in multiple sites occur

usually in unicentric ìocation with subsequent

bony metastases or synchronous or metachro­

nous development. It is a controversy whether

이 논문은 1 990년 6월 22일 접수하여 1 990년 8월 27일 에 채택되었음 Receved June 23 , accepted August 27 , 1990

metachronous osteosarcoma has a multicentric

origin from the outset or represents late

metastasis l- 3 J• However it is known to be diffe­

rent from the synchronous multifocal osteosarco­

ma in regard to the late development of the

second lesion , distribution of the tumors or

prognosis 1.4J. We report a case of metachronous

osteosarcoma in a 6 year old girl with review of

the literatures.

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- Myung .I oon Kim. et al. : Metachronous Osteosarcoma-

Case Report

A 6-year-old g irl was admitted because of

painful swelling of the right wrist for five days

About a 2X2 cm sized. h a rd. tender mass was

palpated on the ulnar aspect of the right wris t.

On the pas t his tory. congenital calcaneovalgus

deformity of both feet was diagnosed at 18

month of age and she was treated with the cor­

rection cast a nd Denis-Brown splin t. All the

laboratory examinations were within normal

limits. The alkaline phosphatase was 175 .4 IU/ L

(normal range; 60- 300 IU / L). Roentgenograms

of the right wrist revealed the mixed sclerotic

and lytic lesions and the periosteal reactions in

the dista l m etaphysis of right ulna (F ig. l-a ). A

whole body bone scan with Tc- 99 m-MDP (Fig.

l - b) showed increased upta ke at th e same site.

A chest film showed no evidence of pulmonary

•

‘ ,

훌 훌

e i

훌 • ‘

a b Fig. l-a. Roentgenogram of the right wrist shows the mixed sclerotic and lytic les ion and the perios teal reaction in the distal metaphys is of ulna b. A whole body bone scan s hows increased uptake at the right wri s t( aITow)

metastasL. An open biopsy of the right ulna was

performed and histology revealed osteosarcoma

Amputation of the right forearm was recom­

mended. but refused by h er p a rents and she was

discha rged without any other procedure. On the

seventh day after discharge. amputation of the

right forearm was performed at another hospita l.

Thereafter she was admitted to our hospital

three times for chemotherapy with vincristine.

adriamycin . cisplatin and the leukocyte &

pla telet combined pheresis

Twenty seven months after the diagnosis of

osteosarcoma . she complained of pain of the

right ankle for 4 days . The blood examination

including alkaline phosphatase was within nor

mal limits. Roentgenograms of the right a nkle

revealed mixed destructive lesions . predomina nt­

ly sclerotic. involving the dist머 metaphysis and

m etadiaphysis of the right tibia (Fig. 2-a). A

whole body bone scan with Tc-99 m-MDP re­

vealed incresed uptake at th e right distal

tibia(Fig. 2- b). A chest film showed no p비mon­

ary metastatic lesions. Biopsy specimen obtained

from the right dista1 tibia showed osteosarcoma.

Curative resection was not performed because of

the refusal of her p a rents. On the last admis­

sion. 10 months after the diagnosis of seconri

tumor. a huge(size; about 20 X 25 cm l. rapidly

growing and bleeding tumor mass was noted at

the right ankle. She also complained of pa in a nd

limita tion of motion of the right ankle. knee and

hip joints. A chest film showed the s mall nodules

in both lungs which was suggestive of p비mon­

ary metastasis. She was disch a rged after the

bleeding control.

Discussion

Since “ multifocal osteosarcom a " was firs t rec

ognized by Silverman51 in 1936. there have been

differen t classifications in osteosarcoma a rising

multiple sites. Lowbeer 61 divided this entity into

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- 大韓放射線훌훌學會註 第 26 卷 第 5 號 1990

했 a b

* • ‘ '}'

‘?~

Fig. 2-a. Roentgenogram of the right ankel twenty seven months after primary lesion shows mixed des tructive lesion , predominantly sclerotic , involving the distal metaphysis and metadiaphysis of tibia. b. A whole body boen scan shows increased uptake at the right distal tibia(arrow)

two types: type 1 , childhood synchronous; type

2 , adult metachronous. Amstutz31 subdivided

into three types; type 1. child-adolescent multiple

almost simu!taneous ; type 2 . adult low grade

malig:tancies ; type 3 . metastatic metachronous

lesions. Mahoney71 devised a new classification

system; group A. child-adolescent synchronous

metaphyseal; group B. adult low grade synchro

nous random; group C. early metachronous(up

to 24 months); group D. late metachronous (af­

ter 24 months).

According to these classifications. there have

been many reports of childhood multifocal

osteosarcoma or unicentric osteosarcoma with

bony metastasis. But the metachronous

osteosarcoma has been rarely reported.

Fitzgerald41 reported 12 cases of multiple

metachronous osteosarcoma in 1973 and de-

scribed that the secondary lesion developed 5 to

163 months after the primary lesion (average; 35

months) . The range of age was 8 to 45 years old.

Tumors tended to be located in the metaphysis

of long bones and showed osteolytic or osteoblas­

tic changes. But one third of the secondary

tumors was dissimilar to the first lesions in the

radiologic features . Enneking and Kagan 1 1 re­

ported that 25 % of metachronous osteosarcoma

arose in the ipsilateral side. But Fitzgerald41

mentioned that there was no way to discriminate

the secondary one from the primaη lesion clini­

cally. radiologically or histologically

Ma ny authors considered multifocal or

metachronous osteosarcoma as the metastatic le­

sion rather than second primary lesion2.3.8.91 and

there has not been any accordance ye t. Generally

primary unict:ntric osteosarcoma metastasize to

spine. pelvis and rarely skull . jaw. sternum. sca­

pula. rib . long bone or sacrum. Skeletal metasta­

sis are more common when the primary lesion

arises in a long bone than when it is within an

axial or girdle bone. and they are also more likely

to occur when there has been recurrence of the

primary lesion 1이 . The metastatic lesion is gener­

ally smaller than the primary one.

In our case. a secondary lesion was located in

the metaphyseal growth center and metadiaph­

ysis of the long bone and was larger than the

pnmarγ lesion and there was no evidence of re­

currence of the primary tumor or metastasis in

27 months after amputation of the right fore­

arm. Belutti 11) suggested the following condi­

tions to establish the multicentric origin of

osteosarcoma: a) absence of previous systemic

bone pathology; b) a negative history of exposure

to radiation; c) absence of lung metastasis or

metastasis in other organ. On her past history.

congenital calcaneovalgus deformity of both feet

was diagnosed at 18 month of age and plain

radiography of both feet was taken at that time.

So we could confirm that there was no previous

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Myung Joon Kim , et al : Metachronous Osteosarcoma -

bone pathology of the right ankel. Pulmonary

metastasis was not noted on the chest film and a

whole body bone scan with Tc-99 m-MDP

showed increased uptake at the right distal tivia.

According to these features , we suspect this case

as the metachronous osteosarcoma rather than

the late metastasis of unicentric osteosarcoma

Pulmonary metastasis was suspected 10 months

after the diagnosis of secondary tumor and it

might be as a result of hematogeneous metasta­

sis from the rapidly grown , secondary tumor.

But Parham21 , Amstutz3 J, Morse l 21 mentioned

that the absence of a pulmonary lesion could not

exclude the metastasis in patients with multiple

bone foci of osteosarcoma. Fitzgerald41 reported

that 5 of 12 patients (42 %) had survived for five

years. And he also considered the metachronous

osteosarcoma might have relatively good prog

nosis as compared with the multifocal osteosar­

coma and unicentric osteosarcoma with bony

metastasis and skip metastasis.

Even though the radiological and histologic

features of each lesion are not different from the

usual osteosarcoma, there seems to be unique in

that secondary tumor develops after adequate

control of the primary tumor and there is no in­

tervening visceral involvement and shows re­

latively good prognosis

In conclusion , adequate treatment of the prim­

ary lesion of osteosarcoma and close follow-up

are very important for the eariy detection and

treatment of the metastatic or metachronously

developed lesion as well as the prognosis .

REFERENCES

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“ skip" metastases in osteo-sarcoma. Clin

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2. Parham DM , Pratt CB , Parvey LS et 려. Chlld­

hood multifocal osteosarcoma; clinicopathologic

and radiologic correlates. Cancer 1985;

55:2653- 2658

3. Amstutz HC. Multiple osteogenic sarcomata­

metastatic or multicentric?; report of two cases

and review of literature. Cancer 1969;

24:923-931

4. Fitzgerald RH , Dahlin DC , Sim FH et 외 Multi­

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5. Silverman G. Multiple osteogenic sarcoma. Arch

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Childhood multifocal osteosarcoma; Diagnosis

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