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Case Report

Coexistent Surviving NeonateTwin and Complete


Hydatidiform Mole
Thomas C. Winter III , Brigit V. BroCk23, Corinne L. Fligner4, Zane A. Brown2

H ydatidiform
with an
moles
incidence
are uncommon.
of approxi-
tidiform
diagnosis.
mole
To
are
our
terminated
knowledge,
after
only
prenatal
a few
measured
tle blood
8.8 x 6.5 x 8.7 cm
flow in this mass was
(Fig. I ). Very
detected using
lit-

mately one in 2000 pregnancies in pregnancies with coexisting fetus and com- either color or power Doppler sonography.
the United States [I]. A twin pregnancy con- plete hydatiditirm mole have been reported The patients ovaries were normal bilaterally
sisting of a complete hydatidiform mole and a to result in a viable or surviving fetus [2-6]. (no theca-lutein cysts were present), and no
coexisting fetus is rare, with an estimated mci- We present what we believe is the first case in ascites or pleural fluid (evidence for hyper-
dence of one per 10,000-100,000 pregnancies the radiology literature of a twin pregnancy stimulation syndrome) was found. The patient
[1]. Coexisting fetus and complete hydatidi- resulting in a healthy neonate delivery with a underwent amniocentesis for karyotype,
form mole differs from classic complete mole normally formed singleton placenta and a which returned 46,XY with an amniotic fluid
and from partial mole in that there are two dis- separate complete hydatidiform mole. This a-fetoprotein level within normal limits at
tinct conceptuses: one conceptus is composed coexisting fetus and complete hydatidiform 1.05 multiples of the median. She had experi-
of a complete molar gestation. and the other is mole pregnancy was monitored by obtaining enced no vaginal bleeding and had no evi-
a normal fetus and placenta [2]. Advanced eight serial sonograms during the time be- dence of hypertension.
technology now available for prenatal diagno- tween diagnosis at 18 weeks gestation and The patient was counseled about the risks of
sis-including serum marker screening. delivery at 36 weeks. gestational trophoblastic disease, preeclampsia,
cytogenetics, and sonography-enables hemorrhage. premature delivery. and choriocar-
the diagnosis of fetus with coexistent mole to cinoma and was offered the option of pregnancy
be made early in pregnancy. The management Case Report termination by evacuation. She chose to con-
of these pregnancies remains controversial. A 24-year-old gravida II para I was referred tinue the pregnancy and was monitored weekly
When compared with singleton (classic) com- to our institution for examination after a triple with blood pressure evaluation and noninva.sive
plete moles, patients with coexisting fetus and screen performed at 16 weeks gestation hemodynamic testing. Platelet count, hemat-
complete hydatidiform mole have higher pre- showed abnormal findings. Her HCG was ocrit, and creatinine levels remained within nor-
evacuation serum human chononic gonadotro- 9.7 1 multiples of the median. and her a-feto- mal limits. [-HCG levels were monitored
pin (HCG) levels (799,590 versus 232, 135 mlii! protein level was 0.91 multiples of the me- closely (Fig. 2). with a peak level at 17.5 weeks
ml) and a greater risk of developing persistent dian, which gave her fetus a one in 47 risk for of 287,000 mIU!ml and a subsequent fall in the
gestational trophoblastic disease (55% versus Down syndrome. Sonography revealed an an- third trimester. She underwent sonographic ex-
14%) [3]. For these and other reasons (such as atomically normal fetus and placenta at I8 aminations every 2 weeks to document ade-
pregnancy-induced hypertension, premature weeks with a coexistent but separate soft-tis- quate fetal growth and to monitor the growth of
delivery, or hemorrhage), most pregnancies in- sue mass that contained numerous small cys- the mass (Fig. 2). The fundal height was consis-
volving a coexisting fetus and complete hyda- tic spaces in the uterine fundus. This mass tently 3-4 cm greater than that expected for ges-

Received June 15, 1998; accepted after revision July 22, 1998.
1 Department of Radiology, Division of Ultrasound, University ofWashington Medical Center, Box 3571 15, 1959 N.E. Pacific Ave., Seattle, WA98195-71 15. Address correspondence to T. C. Winter Ill.

2Department of Obstetrics and Gynecology, Division of Perinatal Medicine, University of Washington Medical Center, Box 356460, Seattle, WA 98195.

3Present address: Swedish Perinatal Associates, 1229 Madison, Ste. 750, Seattle, WA 98104.

4Department of Pathology, University ofWashington Medical Center, Box 356100, Seattle, WA 98195.

AJR 1999;i7245i-453 0361-803X/99/1722-451 American Roentgen Ray Society

AJR:172, February i999 451


Winter et al.
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Fig. 1.- 24-year-old gravida II para I who delivered coexistent normal neonate twin and complete hydatidiform mole.
A, Transverse sonogram obtained at 22 weeks gestation through thorax of sonographically normal fetus and associated normal placenta.
B and C, Transverse (B) and longitudinal (C) sonograms also obtained at 22 weeks gestation show classic appearance of complete hydatidiform mole, located directly
adjacentto normal fetus and placenta seen in A. Part of umbilical cord of normal fetus is seen directly adjacentto complete hydatidiform mole in C (arrow).
D, Photograph of complete hydatidiform mole, with abundant vesicles, no normal villous parenchyma, and no umbilical cord or fetal parts.

tational age. The patient had weekly reactive


nonstress tests from 30 weeks gestation until
250 delivery. Chest radiography and
thyroid func-
tion tests performed early in the third trimester
E 200,000 E
had results within normal limits.
E
150.000 The pregnancy remained uncomplicated
until 36 weeks, when the patient presented to
100.000 I
the labor unit with spontaneous vaginal bleed-
Fig. 2.-Graph shows molar volume
(estimated sonographically by pro- ing and mild cramping. Amniocentesis was
late ellipsoid methods) (crosses) performed, fetal lung maturity was confirmed,
and serum beta human chorionic and labor was allowed to progress. She sponta-
25 30 35 gonadotropin ()3-HCG) levels (cir-
Estimated Gestational Age (Weeks) des) throughout pregnancy. Arrow neously delivered a 2647-g male neonate who
denotes delivery. had an Apgar score of 7!9. Oxytocin infusion

452 AJR:172, February 1999


Coexisting Fetus and Hydatidiform Mole

was begun, and the placenta was delivered in- tion and close follow-up to monitor for poten- datidiform mole was directed at assessing the
tact 6 mm later. The molar mass delivered sep- tial malignant progression. The risk of adequacy detecting any marked
offetal growth,
arately and intact almost immediately after the persistent gestational trophoblastic disease re- change in size of the mole or potential invasion
placenta (Fig. 1). A si.ze-l2 rigid suction can- quiring chemotherapy is approximately 20% of the mole outside of the uterus, and any cvi-
nula was used for curettage, then a Hunter for patients with complete mole and about 5% dence of hyperstimulation syndrome.
curette was used to verify that the endometrial for partial-mole patients. A coexisting fetus In summary, we present a case of the rare
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cavity was empty. The patient and her baby and complete hydatidiform mole, however, entity of a complete hydatidiform mole and a
had an unremarkable postpartum course and presents a dilemma and requires careful con- coexisting fetus that was diagnosed at 18
were discharged on postpartum day 1. Follow- sideration of the risks of continuing the preg- weeks and closely monitored sonographically
up quantitative -HCG levels fell to zero soon nancy, which include progressive gestational and clinically until delivery at 36 weeks. Al-
after delivery and have remained there (Fig. 2). trophoblastic disease in perhaps 55% of pa- though continuation of a pregnancy involving
Pathologic evaluation of the placenta deliv- tients [3], pregnancy-induced hypertension, a coexisting fetus and complete hydatidiform
ered with the neonate showed a discoid term premature delivery. and hemorrhage. mole is rare and is associated with significant
placenta with an eccentric three-vessel umbili- When the sonographic findings are similar medical risks, in this instance both mother and
cal cord and a villous appearance appropriate to those of our patient (a midtrimester, multive- baby did well. Knowledge of the sonographic
for gestational age. The complete hydatidiform sicular, molar-appearing placenta associated appearance and potential complications of
mole measured 12 x 1 1 x 7 cm and consisted with a concurrent normal fetus and placenta). complete hydatidiform mole and coexisting fe-
of a mass of clear, fluid-containing vesicles the diagnosis is essentially an Aunt Minnie tus aids in diagnosis and treatment.
ranging in size from 0.3 to 2.5 cm. Microscop- and is relatively straightforward, even for a rare
ically, avascular hydropic villi with central cis- entity such as coexisting fetus and complete
References
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hyperplasia were seen. No fetal tissue was graphic evidence of a hyperstimulation syn- coexistent fetus. Am J Obstet Gvnecol 1975;122:
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noma was found. The DNA content was theca-lutein cysts and ascites or pleural effu- 2. Bristow RE, Shumway lB. Khouzami AN, Witter
mainly diploid, although approximately one sions. Although the appearance of a partial FR. Complete hydatidiform mole and surviving co-
mole can be similar to that of a complete mole, existenttwin. Obstet GvnecolSure 1996;51:705.-709
third of cells analyzed from one paraffin block
3. Steller MA, Genest DR. Bernstein MR. Lage JM.
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Goldstein DP, Berkowitz RS. Natural of twin
history
pect to find a separate normal-appearing pla-
pregnancy with complete mole and co-
hydatidiform
Discussion centa with the fetus; in addition, the fetus in existing fetus. Obstet Gynecol 1994;83:35-42
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vided into three distinct forms: hydatidiform hydropic degeneration of a second placenta 5. Miller D. Jackson R, Ehlen 1, McMurtiie E. Corn-
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fetus: clinical course of four cases with complete cy-
truens), and choriocarcinoma. These forms ever, no evidence for remnants of a twin preg-
togenetic analysis. GvnecolOncol 1993.50:119-l23
span the continuum from a local degenerative nancy was seen in our patient. Other extremely 6. Vejerslev LO. Clinical management and diagnostic
process to a highly malignant neoplasm [7]. A unlikely possibilities include atypical hemor- possibilities in hydatidiform mole with coexistent
fourth entity, the rare placental-site trophoblas- rhage, degenerating fibroma. or primary pla- fetus. Obstet Gvnecol Sun 1991;46:577-588
tic tumor, has also been described. Hydatidi- cental mass such as chorioangioma. Follow-up 7. McDonald TW, Ruffolo EH. Modern management
ofgestational trophoblastic disease. Obstel Gynecol
form mole has been further subdivided into sonograms would be helpful if the sonographic
Stirs 1983:38:67-83
complete or classic mole, partial mole, and co- findings represented an atypical hemorrhage
S. Snijders R. Farnas M, von Kaisenberg C, Nico-
existing fetus and complete hydatidiform because this would be expected to change sig-
laides K. Ultrasound markers for fetal chromosome
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AJR:172, February 1999 453

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