sarcoma associated with pregnancy

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Sarcoma associated with pregnancy Yutaka Kyodo, MD, Kenji Inatomi, MD, and Tadayuki Abe, MD Shizuoka, Japan A case is reported of sarcoma associated with pregnancy in which the patient underwent a cesarean section with myomectomy and was delivered of a living infant. A pathologic examination indicated sarcoma. (AM J OasTET GVNECOL 1989;161 :94-6.) Key words: Sarcoma, pregnancy Sarcoma is rarely associated with pregnancy. This case report describes a patient with sarcoma in preg- nancy. The pathologic examination revealed well- differentiated leiomyosarcoma. The patient was deliv- ered of a healthy infant. Case report A 29-year-old, gravida 0, unmarried woman with her marriage near at hand requested the examination of antibody titer for rubella on December 23, 1982. On B-scan ultrasonography there were unclear small myomatous nuclei in the uterus (Fig. 1). Her medical history was noteworthy in that she had infantile rheu- matism at 7 years of age and was hospitalized 3 months. On November 25, 1983, at 8 weeks' gestation, on B- scan ultrasonography a tumor three times as large as an adult thumb was detected on the right anterior side ofthe uterus (Fig. 1). Blood and biochemical test results were normal. On May 26, 1984, at 34 weeks' gestation, she had abdominal pain, which was eliminated by analgesics. There seemed to be a partial necrosis in the subserous myoma on B-scan ultrasonography (Fig. 1). On June 20, at 37 weeks' gestation, she underwent a cesarean section with myomectomy because of nonreactivity in a nons tress test and a low estriol level. She was delivered of a living 2640 gm infant with an Apgar score of 9. The placenta was intact and it weighed 700 gm. The membrane was normal. The subserous myoma was the size of a goose egg (80 x 55 mm) and it was soft on the right anterior side of the uterus. There were several holes on the surface of the uterus. Fluid-like pus oozed from the holes. The uterus had several subserous my- omas, but they were left as they were. The subserous myoma of goose egg size was diagnosed as well- differentiated leiomyosarcoma (Fig. 2). On July 6, 1984, she underwent a total abdominal hysterectomy. Bilateral ovaries were intact so they were From the Department of Obstetrzcs and Gynecology, Shizuoka Kosei Hospital. ReceIVed for publicatIOn February 23, 1989; accepted March 3, 1989. Repnnt requests: Yutaka Kyodo, MD, Na/lOnalinstitute for Biolog- iral Standards and Control, Blanche Lane, South MlmllLI, Potters Bar, Hertfordshire, UK EN6 3QG. 94 left alone (Fig. 3). After the surgical procedure she received treatment with Picibanil. a-Fetoprotein, car- cinoembryonic antigen, ferritin, and CA 125 levels were normal before and after the two operations. On August 6, pyelitis was diagnosed. Picibanil was stopped, and she was discharged from the hospital August 19. We have closely watched the course of the disease. Comment Malignant tumor (i.e., malignant ovarian cyst) in pregnancy is rare; it was reported in 1 of 25,000 preg- nancies. 1 Sarcoma associated with pregnancy is rarer than malignant ovarian cyst associated with pregnancy. There are many statistical and pathologic reports with regard to sarcoma, but there are few practical case re- ports that pertain to sarcoma in pregnancy. The sar- coma destructively invaded neighboring tissue and de- generation and necrosis in the tumor center resulted. Sarcoma is well known for its metastasis in the circu- lation of the blood. The purpose of this case report is to describe the resistance to sarcoma cells during preg- nancy. When we first saw the patient she was not pregnant but had subserous myoma in the uterine cavity. After she was pregnant the subserous myoma gradually in- terfered with the developing fetus and was pressed into the anterior segment of the uterus. The sarcoma, which we originally thought to be a subserous myoma, was scattered in the pelvic cavity (Fig. 4). Why couldn't the sarcoma invade the gestational sac? Why couldn't the sarcoma cell in this case pass through the placenta? Sarcomas are very destructive. But the fetal mem- brane was neither damaged nor stained yellow by the sarcoma. It was intact. The gestational sac is made up of three layers: amnion, chorion, and decidua (Fig. 4). Chorion consists of five layers: superficial cells, type IV collagen, type V collagen, type III collagen, and type I collagen. In premature rupture of the membranes, the break in the chorion is thought to be made at the layer of type III collagen by esterase ofleukocytes. 2 But it also has been reported that cancer cells with strong metastatic power secrete large amounts of type IV col-

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Page 1: Sarcoma associated with pregnancy

Sarcoma associated with pregnancy

Yutaka Kyodo, MD, Kenji Inatomi, MD, and Tadayuki Abe, MD

Shizuoka, Japan

A case is reported of sarcoma associated with pregnancy in which the patient underwent a cesarean section with myomectomy and was delivered of a living infant. A pathologic examination indicated sarcoma. (AM J OasTET GVNECOL 1989;161 :94-6.)

Key words: Sarcoma, pregnancy

Sarcoma is rarely associated with pregnancy. This case report describes a patient with sarcoma in preg­nancy. The pathologic examination revealed well­differentiated leiomyosarcoma. The patient was deliv­ered of a healthy infant.

Case report A 29-year-old, gravida 0, unmarried woman with her

marriage near at hand requested the examination of antibody titer for rubella on December 23, 1982. On B-scan ultrasonography there were unclear small myomatous nuclei in the uterus (Fig. 1). Her medical history was noteworthy in that she had infantile rheu­matism at 7 years of age and was hospitalized 3 months. On November 25, 1983, at 8 weeks' gestation, on B­scan ultrasonography a tumor three times as large as an adult thumb was detected on the right anterior side ofthe uterus (Fig. 1). Blood and biochemical test results were normal.

On May 26, 1984, at 34 weeks' gestation, she had abdominal pain, which was eliminated by analgesics. There seemed to be a partial necrosis in the subserous myoma on B-scan ultrasonography (Fig. 1). On June 20, at 37 weeks' gestation, she underwent a cesarean section with myomectomy because of nonreactivity in a nons tress test and a low estriol level. She was delivered of a living 2640 gm infant with an Apgar score of 9. The placenta was intact and it weighed 700 gm. The membrane was normal. The subserous myoma was the size of a goose egg (80 x 55 mm) and it was soft on the right anterior side of the uterus. There were several holes on the surface of the uterus. Fluid-like pus oozed from the holes. The uterus had several subserous my­omas, but they were left as they were. The subserous myoma of goose egg size was diagnosed as well­differentiated leiomyosarcoma (Fig. 2).

On July 6, 1984, she underwent a total abdominal hysterectomy. Bilateral ovaries were intact so they were

From the Department of Obstetrzcs and Gynecology, Shizuoka Kosei Hospital.

ReceIVed for publicatIOn February 23, 1989; accepted March 3, 1989.

Repnnt requests: Yutaka Kyodo, MD, Na/lOnalinstitute for Biolog­iral Standards and Control, Blanche Lane, South MlmllLI, Potters Bar, Hertfordshire, UK EN6 3QG.

94

left alone (Fig. 3). After the surgical procedure she received treatment with Picibanil. a-Fetoprotein, car­cinoembryonic antigen, ferritin, and CA 125 levels were normal before and after the two operations. On August 6, pyelitis was diagnosed. Picibanil was stopped, and she was discharged from the hospital August 19. We have closely watched the course of the disease.

Comment

Malignant tumor (i.e., malignant ovarian cyst) in pregnancy is rare; it was reported in 1 of 25,000 preg­nancies. 1 Sarcoma associated with pregnancy is rarer than malignant ovarian cyst associated with pregnancy. There are many statistical and pathologic reports with regard to sarcoma, but there are few practical case re­ports that pertain to sarcoma in pregnancy. The sar­coma destructively invaded neighboring tissue and de­generation and necrosis in the tumor center resulted. Sarcoma is well known for its metastasis in the circu­lation of the blood. The purpose of this case report is to describe the resistance to sarcoma cells during preg­nancy.

When we first saw the patient she was not pregnant but had subserous myoma in the uterine cavity. After she was pregnant the subserous myoma gradually in­terfered with the developing fetus and was pressed into the anterior segment of the uterus. The sarcoma, which we originally thought to be a subserous myoma, was scattered in the pelvic cavity (Fig. 4).

Why couldn't the sarcoma invade the gestational sac? Why couldn't the sarcoma cell in this case pass through the placenta?

Sarcomas are very destructive. But the fetal mem­brane was neither damaged nor stained yellow by the sarcoma. It was intact. The gestational sac is made up of three layers: amnion, chorion, and decidua (Fig. 4). Chorion consists of five layers: superficial cells, type IV collagen, type V collagen, type III collagen, and type I collagen. In premature rupture of the membranes, the break in the chorion is thought to be made at the layer of type III collagen by esterase ofleukocytes.2 But it also has been reported that cancer cells with strong metastatic power secrete large amounts of type IV col-

Page 2: Sarcoma associated with pregnancy

Volume 161 Number 1

On hov. 7'; . 1

Sarcoma associated with pregnancy 95

On ~c. 12 . 1 Or. ~c. '2 • 1962 lno pregnane), I At 8 eeks' g aWltionl lAl 10 weeks' t'eatat . onl On H3y 26 . I

( Al}4 " ek.· ,,~lnllonl

Fig. 1. Growth and location of sarcoma as pregnancy progresses according to B-scan ultrasonography.

Fig. 2. Subserous myoma of goose egg size was diagnosed as well-differentiated leiomyosarcoma. There are large and small fibrous atypical cells after several mItoses.

lagenase.' The patient's placenta was normal; it had no defect and weighed 700 gm.

Chorion and decidua are part of the endometrium. Decidual cells are said to change the fibroblast cells of endometrium. Fibroblast cells are concerned with fi­bronectin. It is said that fibronectin "catches" carcinoma cells, which enables both leukocytes and macrophages to "kill" them. Fibronectin has an opsonic function against carcinoma cells. There are fibrinoids in the cel­lular space of decidual cells. We believe the role of fibrinoids is to bind both villi and decidua on myo­metrium during pregnancy. Deep in the decidua there is decidual spongiosa, which is tough and has a low response level so sarcoma cells cannot invade the ges­tational sac.

Amnion, chorion, and decidua provide a strong de­fense against foreign invaders; however, they have a low tolerance to inner invaders such as destructive mole

Fig. 3. Total abdominal hysterectomy was performed on day 16 after cesarean section. Specimen has numerous tumors in uterine cavity.

Page 3: Sarcoma associated with pregnancy

96 Kyodo, Inatomi, and Abe

AMNI ON

(;11011111

July 1989 Am J Obstet Gynccol

superficial ......... O~OOO cel ls

collnren .N ... ...... ~ _ ._.- - - ' -

collocen y. ......... -7-=":"=-:-~ ==:-=-coll gen 1lI ......... ~

collBren I ......... ~.w~~~

.................. =:-~~~

DECIl)UA deciduA compnc La .... ~j.

[

~ ... -' fillrifloids

decidua oponCio88"'~

Fig. 4. Sarcoma cells in relationship to structural layers of uterus dunng plegll<lllLy.

and choriocarcinoma. It is natural for villi to invade the myometrium. The human body is helpless with re­gard to internal reproductive invaders. From this case it is evident that both homeostasis and a strong instinct to protect the body from the foreign invader exist in the human body.

REFERENCES I . Terashima Y, Ochiai K. Malignant ovarian tumors associ­

ated with pregnancy. Acta Obstet Gynecol Jpn 1987; 39:145.

2. Kanayama N, Terao T, Kawashima Y, Horiuchi K. Fugi­moto D. Collagen types in normal and prematurely rup­tured amniotic membranes. AM J OBSTET GVNECOL 1985; 153:899.

3. Liotta LA. Tumor invasion and metastasis: role of the base-ment membrane. Am.T Pat hoi 1984; 117:339. '