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No. 5

FIBROSARCOMA

IN INFANTS

& CHILDREN -

Stout

I031

pert in the interpretation of mesenchymal tu- mors, because, in many instances, it is impos- sible t@ determine the exact nature of the tu- mors. The remarkable collection of congenital tumors assembled by Wells33 unfortunately is of no value because the author merely ac- cepted the labels used by the various authors and made no critical analysis. Of 13 cases called either spindle cell sarcoma or fibro- sarcoma, 4 of the patients died of tumor, 5 re- covered, and the result is unknown in the other 4 cases. It would be unsafe to accept any of these as fibrosarcomas. The case reported by Warren and Sommer,32 involving the hand, has already been referred to. There were no metastases. The case reported by Hudson,l7 involving the foot, resembles case 18 in our own series both histologically and because in both cases amputation was necessary. During the short follow-up of 9 months there was no evidence of reappearance of the tumor. Comparable to Hudson’s case is the one re- ported by Dreyfuss.8 It also involved the dor-

sum of the foot, and when the child was 2y2 months old a mid-thigh amputation was done. Dreyfussg called the tumor a spindle cell sar- coma, but it may have been a fibrosarcoma. Shore’s2jpatient is remarkable. The tumor was present at birth in the girl’s interscapular region. It was excised twice and recurred each time. Then it disappeared spontaneously. This tumor I would now classify as a fibromatosis. Hecker’sl5 patient had a congenital tumor in the gluteal muscle removed at the age of 3 months and called a spindle cell sarcoma rich in cells. As the follow-up period was only 3 months and the nature of the tumor uncer- tain, the case is valueless. Hartz and Guerrero Tablante14 reported the case of a congenital tumor of the mastoid region in a baby girl. When she was of 2 months of age, the rapidly growing tumor was excised and the tumor bed electrocoagulated. Seven days after treatment the child died with symptoms of meningoencephalitis. The care- ful histological study justifies the diagnosis of fibrosarcoma. A 3-monrh-old infant had a congenital tu-

No. 5 FIBROSARCOMA IN INFANTS & CHILDREN - Stout I031 pert in the interpretation of mesenchymal

FIG. 3. Case 8. A, This congenital tumor removed from the trapezius muscle has the same general appear- ance in this illustration as the nonmetastasizing tumor shown in Fig. 2. The nuclear markings are slightly more distinct, and the mitotic count was slightly greater but not significantly so. (X230.) B, The recurrence of the tumor. The cells are now distinctly smaller, many are rounded instead of spindle shaped, and a mass of tumor cells have indented one side of a vascular tube. This is probably evidence of loss of differentiation. This patient was the child who died with evidences of lung metastases. (X230.)

  • 1032 CANCEK Sept ern ber-0 ct o bey 1962

YIJI. 15

inor ol the toot. It W~Sexcisecl, re( urrecl

lo-

cally, and an amputation was carried out af-

ter 4 months. Seven months later, the child

died with probable lung metastases. Fahey ancl

Bollingci9 called the tumor sarcoma. It may

have bcen a fihrosai coiiin.

An exircniely interesting casc was reported

by IVilliani~antl Schrum34 as a congenital fi-

brosarconia with widespread metastases in the

skin, SU~CUL~I~~OUS tissues, and viscera. The

boy died on the twenty-wc-ond day after birth.

‘I’his caw was later slutlied by the writer.28

Wc belie\ed it to be an example 01 congenital

gencraliJet1 fibrornatoscs with multiple foci

and not a metastasizing fibrosarcoma.

‘I’his intomplcte survey oE probably congen-

ital fibrosarcomas show how completely frus-

trating the data concerning this subject have

proved to be. But it seems significant that in

only 1 cnse. that oE Fahey and Bollinger,9 was

there ekitlenc e of distant metastasis in the

lungs, aiirl in that case the diagnosis of fibro-

sarconia is yuestionablc.

Fibrosarconias sometimes develop in the

deepcr tissue? ancl the viscera of children. A

numbcr have been described that have in-

volved the bronchi and lungs.

I,c~is’~~

cabe was in the bronchus of

il

14-

year-old bo) . Following bronchoscopic removal

and radiotherapy, he ieminerl well lor 2%

years. Feldman’slo casc 1 was in an 11-year-old

girl. After segmental resection of the lung

tumor in the left lower lobe, she remained well

for more than 3 years. In the photornicro-

graph, the tunior looks like a fibrosarcoma.

Donoghue ct al.’s7 patient was a girl 5% years

of age. She had an endobronchial fibrosarcoma

treated by right pneumonectoniy. She was well

after 3 months but could not be traced after

that. Curry ancl Fuch’sG patictit was ;I 15-year-

old girl who coughed up ;F fibrosarconia. She

received no treatnient and was well 4 years

later. Holinger et al.’slo patient was a 51/-year-

old boy with a bronchial fibrosarcoma. The

right middle and lower lobe5 were resected.

He recovcred itom the opelation, hut there

was no follow-up. In the case ol Gerbasi et al.lR

the child died with metastases in the opposite

lung. The 2-year 9-month-old boy was treated

by lobectomy antl radioactive cobalt therapy.

Although called fibrosarcoma, this case can-

not be accepted be( ;i uw

the pliotomitrographs

leave the nature of the tumor uncertain, and

there is no report of proper histological investi-

gation. Hence reporled fibrosarcoma of the

lung in children has not proved to be any

more nialignaiit than fibros;ircoma elsewhere.

Fi brosarcoina has occahiia lly becn rcportcd

FIG 4. Case 9. A, The gross appearance of thc bisected tumor in the vastiis mcdialis
FIG 4. Case 9. A, The gross appearance of
thc bisected tumor in the vastiis mcdialis
niu\clr. B, ‘The illustration shows cells of
irietlircin siie and spindle shape arrangement
in inteilacing bands. In other parts of this
tiinlor. them were larger and smaller cells.
In some aicas, reticulin fibers were wiapped
about each rell; in otheis they were sparse.
Mitoses average 12 in 50 high power fields.
‘11ii$ tumor recurred and was re-excised. In
spite 01 tlie suggestion\ of possible malig-
nanc\, the tliilcl was a 5-year cure. (~230.)

No. 5 FIBROSARCOMAIN INFANTS& CHILDREN- Stout 1033

No. 5 FIBROSARCOMA IN INFANTS & CHILDREN - Stout 1033 FIG. 5. Case 13. The cells

FIG.5. Case 13. The cells of this tumor of the ton- sillar fossa have the usual interlaced arrangement and vary in size from medium to slightly smaller. Reticulin fibers surround each cell. The mitotic rate is negligi- ble, and there is little to distinguish this tumor from

many of

(~230.)

the others. In spite of this, the child died.

in the upper respiratory tract of children. Bah- gat1 described a 13-year-old girl who had a mass filling the naso- and oropharynx and en- croaching on the laryngopharynx. It was ex- cised, and after 3 months there was no evi- dence of recurrence. Berard and Sargnon2 described a tumor that filled the nasopharynx and the maxillary sinus and invaded the orbit, displacing the globe, and made the palate bulge into the mouth. It was excised, and 2 radium treatments were given. The patient was well 20 years later. This seems more descriptive of osteofibroma (fibrous dysplasia) than of fibrosarcoma. Cabot’s* patient (case 16302) was a 6-year- old child in whom the tumor started in the temporal region; it was excised and treated by X ray and radium but recurred in the cheek and extended to the maxilla, maxillary sinus, malar, temporal, and ethmoid bones, and the ethmoid sinus. It was called a fibrosarcoma but did not metastasize. Fibrosarcomas have been reported in the retroperitoneum of children by Melicow22 and

by Snyder et a1.,26 but since these authors have given no photomicrographs or histological de- scriptions, the cases cannot be evaluated. Gardner and Turner11 described a fibro- blastic tumor that arose in the choroid plexus of the lateral ventricle of a 3G-year-old child. It was excised, and 9 months after operation the child was symptomatically improved. From these case reports concerning presum- ably acceptable juvenile fibrosarcomas it is found that, of 61 patients, 29 were followed for various periods and did not have evidence of metastasis, 80 were not followed but pre- sumably did not show evidence of metastasis at the time of reporting, and only 2 gave evi- dence of metastases. It is quite true that sev- eral of the tumors infiltrated extensively and incomplete removal resulted in recurrences that led to amputation and sometimes were directly responsible for the death of the pa- tient, but, since they were not known to have metastasized, they were potentially curabsle. Moreover, 2 of the tumors disappeared, I af- ter radiotherapy and 1 spontaneously. It is quite evident that so-called juvenile fibrosar- coma differs very little from juvenile fibro- matosis, and it becomes of great importance to learn how to recognize the ones that are po- tentially metastatic from all of the others. Since sections of the reported tumors that metasta- sized are not available to the writer, it will be necessary to glean what information is possible from the new cases to be reported from this institution.

PERSONALCASES

Etiological Features. Sex. Fourteen of the patients were boys, 7 were girls, and the sex of 2 is unknown. Age at Onset. As has sometimes been the case in other mesenchymal tumors, a majority first manifested themselves in either the first or third quinquennia. The onset of 11 cases was in the first quinquennium (birth to 5 years), and 4 of these were congenital. Five were first noted in the second quinquennium (ages 6 to 10 years), and 7 in the third (11 to 15 years). Site of Tumor. The tumors were widely scattered: head and neck, 7; trunk, 4; upper extremity, 4 (arm 1, forearm 1, hand 2); lower extremity, 5 (thigh 1, leg 1, foot 3); oral cavity, 3 (tongue, gum, pharynx): bronchus, 1; spinal meninges, 1. None of the tumors was primarily in the skin. Intramuscular tumors included

I034

CANCEKSept embt:r-October 1962

Vol. 15

‘1

in

the external soft

tissues and

1

in

the

t.ongue. One tumor was attached to the perios-

teirni of the scapula.

l+tr t Ii ologicu I Findings.

Gross Description.

--

__

Reference to Table 1 will show that. in cases in Case p&s

Ses

which the size was known, most of the tumors

110.

no.

Pt.

were small. However, it must be realized that

1

29461

?

~.hc.none is dealing with infants, a tumor that

2

30949

F

1% odd be considered small or not very large

3*

35430

M

iti ;in adult would assume tiiore formidable

4

39421

?

~xoportions.For example, in case 18, the child

was born with a tumor enlarging arid deforrn-

ing an entire foot. Compared with the size of

5

43104

M

thc LhiJd, the 9x8~7-cm. lumorr’ appeared

C~OI’I~~US. The consistency of the tumors

6

462 1 7

47245

M

varied; soinc were hard, others soft; there was

7

49176

M

c onipletc variabilily among them. They were

8

50572

M

seltlom painful, and the nature of the tumor

51312

coultl not bc deduced from the clinical ex-

9

51944

F

aniiii~iti~~~i.

hlicroscopic Desa iption. The great major-

ity of the tumors were composed of spindle-

5h;ipetl cells with a tendency to be arranged

in sinuous bands or cords that interlaced. The

11

54311

F

elongated cell bodies tended to be pointed,

.inti the nutlei conformed to the shape of the

12

58330

F

body and were also pointed. There were no

in t r<icc.1 I u I <tr fibrils, b 11 t reticulin

fibers coni-

13

60212

F

nioiily surrounded each cell and there were

also often elongated reticiilin fibers parallel-

14

60846

kl

ing the long axes of the cells (Fig. 6A and B).

15

61366

M

Very occaGonally the reticulin fibers were not

wrapped about each individual cell but were

only prscly scattcred among them. Since this

16t

62230

ill

was found in cases 5 and 8, the 2 cases known

17

63326

M

to have inetastasixd, it is probably an indi-

18

6.3875

M

c;ttion of increased malignancy (Fig. 1K and

11). The usual length of the cells is shown in

Figs. 2 and 6.4, and this length is labeled

19

61512

M

“nictliuiii” in the cable. ‘Tumors were some-

20*

64713

F

times composed of cells either larger or smaller

than “medium”

.SU, 7, and 8A). In true

21

64718

M

human fibroblastic tumors, real giant cells

with either single or multiple nuclei are hardly

22

ti5057

M

ever found. If any appreciable number of gi-

xnt cells are found in a rpindle cell tumor, it

is probably ci ther a liposarcoma, a rhabdomyo-

23

654.59

F

PERSONAL CZSES

_-

I’t.

4t

age, yr.

-_

At

onset

treat.

?

5

5

74/12

11

Birth

1-14 /12

3/12

10

2-5/12

8

Birth

24/12

11

2-6/12

8-6/12

11/12

2-6/12

12

13

-I_

Site turn.

Scalp

Scap. reg.

Bronchus

Gastpc-

Iiemius

muscle

Pal rn

Over lacr.

Sac

Forcarm

Trapezius

muscle

Vastus

niedinlis

muscle

Foot

,

3

6/12

a

Pcrios-

teum;

scap.

Tongue

12

4

15+

12-1/12

6

16

7

?

Birth

7

12

1 day

Tonsillar

fossa

Pa1 m

Lymph

iiodc at

lat. dorsi

muscle

Mast. reg.

Axilla

Foot

1-5/12

11

14/12

11-2/12

?

11

9

8

12

10

Nas. reg.

Gum;

maxilla

Epidural;

L-2 & 3

Ihl toid

reg.

Sole foot

sarcoma, or a leiomyosarcoma. All of the fibro-

sarcomas infiltrated their environs. If the nu-

*The patient was a Negro.

c-lcus resembled the nuclei of thc cells in fibro-

niatoses, it was recorded as “no anaplasia.” If

the nuclear markings were more accentuated,

it was recorded as “slight or definite ana-

plasia.” In definite anaplasia, the cell body us-

ually was well defined. Often in fibromatoses

the nucleus is not too clearly defined. It was

hoped that the relative number of mitoses

might have some relationship to malignancy.

Therefore in each case their number in 50 high

power fields has been recorded. Apparently

this has not been a reliable criterion, and one

is forced to assume that it is simply an indica-

  • 1056 CANCERScptem ber-Octobel- 1962

1'01. 1.5

CANCER Scptem ber-Octobel- 1962 1'01. 1.5 Frc. G. Case 14. A, This is the recurrence

Frc. G. Case 14. A, This is the recurrence of the tumor in the palm. The cclls are of medium sizc. have thc usual

iriter1;icccl appi'armce, and iriGItrate thc III~ICU~~IICOIIS

fat. (~230.)13, The silver reticulin stain shows fine retic-

itlin tilwrs siirroui~diiigeach cell. aud some paralleling the long axes of the spintlle-shapedcells. (~230.)

1 pait oi a s)novial sarcoma may reveal only

the fibi o~arcomatouselcrnent. He is unable to

rccogniie this when it occurs antl knows of

no~hingshot t of tiswe culture tliat could cie-

Lide thc choice. He would prefer to err by

calling ;I syiiovial s'ircoma a fibrosarcoma, be-

cauw apparently the tumor is more apt to

display thc less malignant characteristics of a

fibrowrcom;i cwn if it should be a disguised

synovial 5arcoma.

iliiother cutrenicly difficult problem has

becii to try to decide where to draw a dividing

li tie be tu een fib1 onia~o5cs,ind fibrosarcomas.

In selecting this group of cases, fibroblastic tu-

mors with a marked degree of cellularity were

arbitrarily chosen. The writer is well aware

that this is far irom exact since obviously the

tei in "marked" will bc differently interpreted.

Actually it has very little significance, since,

in children, the proportion of fibroblastic tu-

mors that will metastasize is very small. This

que5tion will be further discussed under indi-

cations for treatment.

Results of Treaiment. It has been possible

for the majority of those who consulted the

writer about these tumors to furnish him with

reliable follow-up data. At the timc of writing,

15 patients were reported lree lrom evidences

of tumor. Six were well from 5 to 12 years af-

ter last treatment and 9 others from 1 to 5

years. Onc was well for 8 months and then was

lost to iollow-up. Onc (case 23) was well 22

months alter excision with iecurrcnce in the

scar. The child with tumor involving the spinal

meninges (case 21) was syrnptoiriatically im-

proved 3 months after treatment by radio-

therapy. Three children died: 2 (cases 5 and 8)

were known to have had rnetmtasis, antl the

cause of death of the third (case 13) is not

known. Two patierits were lost LO follow-up.

One child (case 15) had a tumor involving the

latissimus dorsi muscle with involvement of an

axillary lymph node. Whether this was due to

metastasis or due to direct invasion is not clear,

but the child was well 3 years after treatment.

Three children had extremities amputated

(cases 4, 18, and 22) but are not known to have

had any metastases.

Drscr fswm

As a result of studying the cases of lesions

called fibrosarcomas in children reported in

the literature and a further study of the pres-

  • 1038 CANC,ERSeptem bey-October 1962

Vol. 15

CANC,ER Septem bey-Octo ber 1962 Vol. 15 FIG. 8. Caw 18. Yibrosarcoma of foot. A,

FIG. 8. Caw 18. Yibrosarcoma of foot. A, Characteristic field showing the relatively small spindle-shapcd cells occasionally arranget! in vague bundles. The edcma and the many infiltrating lymphocytes confuse the picture. (~230.)B, Silver retirillin stain showing that many of the cells are wrapped ahout by the very delicate reticulin

lihcrs. (~230.)

tissue tumors in children is to remove them

piecemeal or in a single piece but close to the

tumor :is if it were a cyst. ‘These fibrous tu-

mors are never encapsulated and always in-

filtrate to some degree. If, after biopsy or in-

complete excision, the tumor proves to be a

fibrosarcoma, one should not sacrifice extrem-

ities without making every attempt to achieve

a cure by less drastic measures. ‘The fact that

the chances of metastasis are SO remote in a

given case should influence the surgeon’s judg-

ment in planning treatment.

CASEREPORTS

Case 5. When this Brazilian white boy was

10 years old, a tumor was noted in the thenar

region of the right hand. When he was 11

years old, the tumor occupied the entire the-

nar region and extended into the thumb as

far as the distal phalanx. A wide local exci-

sion was carried out because the tumor invaded

muscle. A chest roentgenogram at this time

was negative. Eights months later, an axillary

node dissection was carried out. One among

44 nodes had a metastasis. Seventeen months

after this axillary opcration, the patient died

at home, and no autopsy was obtained. His-

tologically, there was nothing to suggest ma-

lignancy in he hematoxylin and eosin-stained

sections. The sections stained with Laidlaw’s

stain showed that reticulin fibers were not

wrapped about each cell. The metastasis in

the lymph node showed exactly the same pic-

ture (Figs. lA, B, C, and D).

Case 7. This 8-year-old boy developed a

painless mass on the anterolatcral aspect of

the upper left forearm. Six months after its ap-

pearance, a 2-cm. circumscribed tumor was re-

moved from the subcutaneous tissue. After 4

months, the area was re-excised because of a

recurrence. Six years and 3 months later, the

boy was well and free from any evidence of

tumor. The tumor was well differentiated and

quite vascular. Mitoses were rare, and the tu-

mor was on the borderline between a well-

differentiated fibrosarcoma and a fibromato-

sis (Fig. 2).

Case 8. This boy was a difficult breech de-

livery, and the neck was supposed to have been

injured during it. A lesion developed in the

trapezius muscle. 4t the age of 11 months, the

growth was removed piecemeal from the mus-

cle. In less than 3 months, there was a re-

currence, and roentgenograms at this time

showed that the lungs were riddled with metas-

KO. 5 FIBROSARCOMA IN INFANTS 8c CHILDREN - Stout 1039

tases. In 3 more months the child was dead. The original tumor looked in part like a dif- ferentiated fibromatosis but had areas in which the tissue was more cellular, the nuclei suggested anaplasia, and some cells were rounded instead of spindle shaped. Recurrence showed a reproduction of the more active part of the primary tumor (Fig. 3A and B).

Case 9. At the age of 2% years, this girl was found to have a 5-cm. tumor embedded in the

muscles of the right

thigh. A local excision

was carried out, but the tumor recurred. Af- ter 8 months, the recurrence was excised. There was no further reappearance of the tumor, and the child was reported well 5 years and 5 months after the second operation. The closely packed spindle cells showed some nu- clear anaplasia and a considerable number of mitoses and gave the impression of malignancy (Fig. 4A and B).

Case 13. A 12-year-old girl was found to have a growth in the left tonsillar fossa 5 weeks af- ter tonsillectomy. When the tonsils were re- moved, a 1-cm. nodule was exposed in the left fossa. In 5 weeks, it had reached a diameter of 4 cm.It was removed piecemeal. It has been possible to learn only that the child died 19 months later. Whether there were metastases or recurrence is unknown. Histologically, the tumor was quite cellular but with most of the nuclei appearing normal or with only a slight suggestion of anaplasia and very few mitoses

(Fig. 5).

scribed, 3.2 cm. in diameter, and was not re- moved intact but broken into during the op- eration. In spite of this, there has been no evidence of recurrence or metastasis during the 3 years that have elapsed since the op' eration. This tumor had a mixture of cords of spindle-shaped cells and undifferentiated rounded and spindle-shaped cells. Mitoses were frequent, and some cells appeared anaplastic (Fig. 7). The tumor was partly surrounded by lymphoid tissue, suggesting a compressed lymph node.

Case 18. This boy, a first child, delivered normally after an uneventful pregnancy, was born with a large tumor mass involving the entire right foot anterior to the heel. A mid- leg amputation was carried out the next day. During the 2 years that have elapsed since the operation there has been no evidence of recurrence or metastasis. During this period, a tumor of unknown type was removed from the back. The tumor of the foot, measuring 9X8X 7 cm., involved all the soft tissues of the foot and almost obliterated the toes. There was extensive necrosis and ulceration of the tumor on the plantar surface. The tumor was com- posed largely of rather small spindle-shaped cells that sometimes formed bundles but else- where had no definite arrangement. In many areas, inflammatory cells and engorged capil- laries confused the picture. Very fine reticulin fibers surrounded each cell. Mitoses were rare (Fig. 8A and B).

Case 14. A four-year-old Jewish boy was found to have a swelling of the right hypothe- nar eminence. Two years later, a tumor was excised and recurred almost at once. Its origi- nal size was not recorded, but the tumor was reported as a dermatofibrosarcoma protuber- ans. The recurrence was excised about 3 weeks after the first operation. Two years and 9 months after the second operation the child was well and free from evidence of tumor. The recurrence appeared relatively well differenti- ated, with reticulin around each cell, no ana- plasia, and a moderate number of mitoses (Fig. 6A and B).

Case 15. This 16-year-old boy first noticed a

swelling in the latissimus dorsi muscle 15 a. lateral to the osterior mid-line at the level of

the twelfth

Borsal vertebra. The lesion was

painless and was first noticed at stretching ex-

ercises during football practice. It was circum-

SUMMARY

A study of available reports of probable ju- venile fibrosarcomas in the literature and of 23 personal cases suggests that this is an un- common tumor type in children. It can de- velop in the soft tissues and rarely in the vis- cera in many parts of the body. The tumor is difficult to distinguish from the fibromatoses and seldom metastasizes, probably not more than once in every 13 cases. It has not been possible for the writer to be able to distinguish those that will probably metastasize from the others, although certain features are sugges- tive. Since all fibrous tumors in children infil- trate, the indication for treatment is directed entirely toward complete removal of the local lesion. If this is accomplished, almost all cases will be cured.

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1933.

3. BICK,E . M.: Fibrosarcoma of extremitics. Ann.

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I040

CANC,ERSeptembel--October 1962

Vol. 15

  • 4. Cnwr, C. K.,E d.: Case 16902. In Case rccords of

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COLLINS,

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W. E.: Fibrosarconia

Of p1;iiitar

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.4nn.

.I. Cancer 40: 465-470, 19.10.

G. (;I'KKY,

J.

I., and FUCHS,J. E.:

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7.

I)o~ocr-iu~,

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8.

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LEWIS,I.: Sarcoma of bronchus. Proc. Roy. SOC.

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MALLORY,

T. B.,

Ed.:

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PACK,G. T

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FAHEY,J. J.,

and

J.

BOLLINWR,

A.:

Congenital

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FH.DMAN,P. A:

Sarcoma of

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report

of

3

rases. Brit. J. 'I'uberc. 51: 331-334, 1957.

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PRIOR,J. T., and Srsso~,B. J.: Dermal arid fascia1

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curring

SHORE,B. R.: Spontaneous cure of congenital re-

connective-lissoe

tumor.

A7n.

.I.

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736-739, 1936.

11.

(.~wNF,R,

W.

I,, and

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