Open Med.

2019; 14: 843-846

Case Report

Chengying Zeng, Yanbi Chen, Lijuan Zhao, Bo Wan*

Partial hydatidiform mole and coexistent live

fetus: a case report and review of the literature
https://doi.org/10.1515/med-2019-0098 tinct disease processes with characteristic cytogenetic,
received January 20, 2019; accepted September 30, 2019 histological, and clinical features [2]. Partial hydatidiform
moles (PHM) originate from dispermic fertilization of a
Abstract: Twin pregnancy of a hydatidiform mole with normal haploid oocyte that generally generates a trip-
a coexistent live fetus is very rare, and complete molar loid set of chromosomes. A complete hydatidiform mole
pregnancy is involved in most cases. A partial molar preg- (CHM) contains 46 diploid paternal chromosomes. In PHM
nancy almost always ends in miscarriage due to a trip- pregnancy, the fetus can develop but is malformed and
loid fetus. Here, we report a case of a 32-year-old Chinese non-viable, whereas no fetus develops in CHM pregnancy.
woman with ultrasound diagnosis of a partial molar Most twin pregnancies with hydatidiform mole are CHM
pregnancy. Amniocentesis suggested mosaicism, but the pregnancies with a normal fetus and placenta [3]. Reports
fetus was morphologically normal. The woman chose of PHM pregnancy with a live fetus are extremely rare
to continue the pregnancy after fully understanding the because triploid fetuses tend to die in the first trimester
risk. The infant was delivered prematurely, and the pres- [4-7].
ence of a large single placenta with molar changes. The To our knowledge, only 19 cases of singleton, PHM
baby’s peripheral blood chromosomes were diploid, and pregnancy have been reported, five of which continued to
the pregnant woman had no serious complications. The successful delivery of a baby with diploid karyotype [8].
diagnosis, management, and monitoring of this condi- In this paper, we present a case of a PHM pregnancy
tion will remain challenging because of its rarity. Partial coexisting with a diploid fetus that was later live-born.
hydatidiform mole combined with pregnancy can result in The woman presented with vaginal bleeding and a seem-
delivering of a normal fetus and live birth under proper ingly huge placenta; amniotic fluid karyotyping revealed
management. pseudomosaicism.

Keywords: Partial molar; Normal viable fetus; Prenatal

diagnosis; Pseudomosaicism; Case report
2 Case report
On 17th May 2018, a 32-year-old Hui Chinese woman,
gravida 3, para 2, presented with vaginal bleeding not

1 Introduction accompanied by uterine contractions at 22 weeks of ges-

tation in the Department of Gynecology and Obstetrics of
The Third Affiliated Hospital of Southern Medical Univer-
Twin pregnancy with hydatidiform mole and a coexist-
sity. The patient had no family history of gynecological or
ent live fetus is a very rare condition occurring in 0.005%
obstetric diseases and denied having taken sex hormone
to 0.001% of all pregnancies [1]. Hydatidiform moles are
drugs. Ultrasonography (US) revealed a fetus with normal
categorized into complete and partial types that have dis-
anatomy and an appropriate amniotic fluid volume. An
abnormally thickened, single, large multicystic placenta
with placenta previa was also observed. A provisional
*Corresponding author: Bo Wan, Department of Obstetrics and Gyne-
cology, The Third Affiliated Hospital of Southern Medical University,
diagnosis of hydatidiform mole coexisting with a live fetus
Guangzhou 510630, Guangdong, China, Tel: +86 13660513710, Fax: was proposed.
+86 2062784383, E-mail: [email protected] The serum β-human chorionic gonadotropin (β-hCG)
Chengying Zeng, Yanbi Chen, Lijuan Zhao, Department of Obstetrics was 169,200 mIU/mL. The patient underwent amniocen-
and Gynecology, The Third Affiliated Hospital of Southern Medical tesis, and the amniotic fluid karyotype revealed was:
University, Guangzhou 510630, Guangdong, China
46, XN[51]/92, XXNN[34]. The results of quantitative flu-

Open Access. © 2019 Chengying Zeng et al. published by De Gruyter. This work is licensed under the Creative Commons Attribution 4.0
License.
844    Chengying Zeng et al.

orescence-polymerase chain reaction (QF-PCR) showed blood loss was approximately 400 mL. Histopathological
that amniotic fluid cells were chimeric with triploid and examination of the placenta showed a mixture of large,
diploid, and the chimeric ratio of triploid was 10%. False edematous, irregular villi, combined with small nor-
mosaic phenomenon in amniocentesis had not been ruled mal-sized none dematous tissue. Trophoblastic hyperpla-
out so we suggested performing an umbilical cord blood sia of the villous surfaces was limited and focally reminis-
puncture or amniotic fluid cell fluorescence in situ hybrid- cent of PHM (Figure 1B).
ization (FISH) review to determine whether it was true The postpartum karyotype of the neonate revealed
mosaicism or pseudomosaicism, but the patient refused both 46, XX. The postoperative course was uneventful,
further examination because of economic reasons. The and she recovered well and was discharged on Day 5 after
patient was further informed of the risk of preterm birth the cesarean section. β-hCG concentration was 35,927
and trophoblastic disease, but for religious reasons, she mIU/mL on the first day after the discharge; by the second
chose to continue the pregnancy. week it was 2,774 mIU/mL, 420 mIU/mL by the fourth
On 24th June 2018 (29 weeks and 3 days of gestation), week and dropped to 0 by the tenth week. Four months
the patient experienced increased vaginal bleeding and after the birth, the infant weighed 4.2 kg and had a height
abdominal pain and was admitted to hospital. Upon of 53 cm. The baby is currently growing well.
admission, the serum β-hCG was 153,590 mIU/mL, and the
hemoglobin was 9.5 mg/dL (Mild anemia). The remain- Ethical approval: The human research part of our study
ing physical examination and laboratory workup (blood complied with all relevant national regulations, institu-
pressure, serum transaminases, thyroid hormones, blood tional policies, and was conducted in accordance with
platelets and creatinine, with urinalysis, and chest X-ray the tenets of the Helsinki Declaration. The present inves-
examination) were normal. The baseline fetal heart rate tigation was approved by the authors’ institutional review
was 140 bpm. The fetus was sensitive to non‐stress test. board or an equivalent committee.
The woman received intravenous magnesium sulfate 1 g/h
for 48 h and dexamethasone 6 mg q12h for two days. Informed consent: Informed consent was obtained from
Uterine contractions increased on the third day of all individuals included in this study.
hospitalization. The patient and her spouse were coun-
selled on the risks and implications of the diagnosis.
Under combined spinal-epidural anesthesia, a low trans-
verse incision cesarean section was performed and a 1050
3 Discussion
g, 38-cm long live female baby was delivered with 1, 5, and
Here, we report a case of a 32-year-old Chinese woman
10-minute Apgar scores of 9, 10, and 10, respectively. The
with a partial molar pregnancy and live birth of a diploid
placenta, which was large and hydropic, with necrotic
infant.
debris, was recovered manually (Figure 1A). The estimated

Figure 1: (A) The placenta was large and hydropic, with necrotic debris; (B) The trophoblastic hyperplasia of villous surfaces was limited and
focally reminiscent of partial hydatidiform mole (Magnification was ×100).
 Partial hydatidiform mole and coexistent live fetus   845

Hydatidiform mole has been known since the time sary to fully inform the pregnant woman of the possible
of Hippocrates and is characterized by a wide spectrum maternal and fetal complications, such as preeclamp-
of presentations and rare spectacular complications [9]. sia, hyperthyroidism, vaginal bleeding, and theca lutein
CHM consists of a diploid set of 46 chromosomes, and ovarian cysts. The probability of postpartum development
all genetic material is of paternal origin, with no fetal into persistent trophoblastic disease is high. In cases of
structure; it is noteworthy that the risk for trophoblastic PHM, only few villous vesicular changes occurred; the cel-
sequelae (15%–20%) is higher than that of pregnancy with lular proliferation was nourished, 90% of the fetal chro-
PHM (<5%) [10]. PHM pregnancies result from fertilization mosome karyotypes were triploid, and most pregnancies
of an apparently normal ovum by two sperm, giving rise ended with an abortion and fetal death. The probability
to a triploid karyotype (69, XXY). Three types of molar of postpartum development into persistent trophoblastic
pregnancy concomitant with a normal live fetus have disease was established to be 4%, which is much lower
been identified so far, of which twin pregnancy with one than that for CHM; hence, chemotherapy is rarely needed,
normal fetus having a normal placenta and another CHM and generally no metastasis occurs [16, 17].
is the most common; the second type is twin pregnancy In the present case, the patient had slight vaginal
with a normal fetus and placenta and another PHM, and bleeding during the second trimester of pregnancy. Her
the third and most uncommon occurrence is a singleton blood hCG dropped to normal one month after the deliv-
normal fetus with PHM pregnancy [8]. ery, and postoperative placental pathology confirmed the
Ultrasound is the main method for hydatidiform diagnosis. With the diagnosis of PHM we first considered
mole diagnosis [11]. The ultrasonographic appearance of that the pregnancy should be terminated. However, this
PHM is usually honeycomb-like echo in the placenta; the case showed that PHM can result in a normal fetus and
boundary between the normal placental tissue and the live birth under proper management. Therefore, although
honeycomb echo is not clear, and most fetuses are dead the incidence of this condition is very rare, this case is
or malformed [12]. Few prenatal ultrasounds of PHM are important because recognizing and diagnosing PHM is
structurally normal [4]. In our case, at the 22nd week, the vital for patient care. Moreover, it should be considered
ultrasound revealed that a part of the placenta was hon- and looked for in patients presenting with pre-eclampsia.
eycomb-like with a cyst-like echo, but the coexisting fetus Interventional prenatal diagnosis was performed by ultra-
showed no abnormal structures. Amniotic fluid karyo- sound-guided transabdominal chorionic villus biopsy
typing indicated that the fetus was a diploid/tetraploid and amniocentesis and combined with interphase FISH
chimera; QF-PCR confirmed diploid/triploid chimerism. to determine fetal karyotype [18]. During this pregnancy,
Because ultrasound showed a normal fetal structure, we the condition of both mother and fetus was monitored and
considered the possibility of pseudo-chimerism of the followed up strictly according to the principle of hydatidi-
amniotic fluid. Further, the maternal and fetal conditions form mole follow-up.
were monitored according to the patient’s wish. There The termination of pregnancy with hydatidiform mole
was no significant increase in serum hCG, and the fetal depends on the gestational duration and disease condi-
growth index was normal. Finally, a live baby with normal tion; in early pregnancy this is often associated with com-
appearance and karyotype was delivered. PHM was diag- plete curettage of the uterine cavity. The results of the use
nosed by postoperative placental pathology. of intra-amniotic injection of rivanol and intravenous oxy-
The hCG level in CHM was significantly higher than tocin or cesarean section in the second trimester of preg-
that in PHM. Less than 10% of PHM patients were pre- nancy are controversial [19]. Caesarean section is recom-
viously found to have hCG greater than 100 KU/L [13]. mended to deliver a live fetus with hydatidiform mole, as
However, a high level of hCG was detected in the present repeated uterine contraction increases the possibility of
case, which might have been related to the large propor- squeezing of the hydatidiform tissue into the abdominal
tion of hydatidiform mole tissue in the placenta. cavity, leading to increased risk of pulmonary embolism.
Pregnancy complicated with hydatidiform mole is Due to placenta previa and premature labor, the patient
usually terminated immediately after diagnosis [14]. Cur- enrolled in this study chose lower uterine segment cesar-
rently, it is well understood that most CHM fetuses develop ean section. Oxytocin was used after the rapid removal of
normally, and thus women can choose to continue preg- the grape tissue during the operation. The uterine con-
nancy with appropriate support [15]. However, it is neces- traction was good, with little bleeding.
846    Chengying Zeng et al.

4 Conclusion [8] Kawasaki K., Kondoh E., Minamiguchi S., Matsuda F., Higasa
K., Fujita K., et al., Live-born diploid fetus complicated with
partial molar pregnancy presenting with pre-eclampsia,
In this report, we have described a rare case of partial maternal anemia, and seemingly huge placenta: A rare
hydatidiform molar pregnancy coexistent with a live case of confined placental mosaicism and literature review,
fetus with pseudomosaicism. This case shows that under J Obstet Gynaecol Res, 2016, 42, 911-917; doi 10.1111/
jog.13025
proper management, PHM combined with a live fetus can
[9] Cagayan M.S., Hydatidiform mole and its complications:
result in live delivery of a normal infant. review of patient profiles and management at the university
of the Philippines-Philippine General Hospital, J Reprod Med,
2014, 59, 235-240
Acknowledgements: None [10] Lurain J.R., Gestational trophoblastic disease I: epidemiology,
pathology, clinical presentation and diagnosis of gestational
Conflict of interest: The authors declare that they have trophoblastic disease, and management of hydatidiform
no conflict of interests. mole, Am J Obstet Gynecol, 2010, 203, 531-539; doi 10.1016/j.
ajog.2010.06.073
[11] Ronnett B.M., DeScipio C., Murphy K.M., Hydatidiform moles:
ancillary techniques to refine diagnosis, Int J Gynecol Pathol,
References 2011, 30, 101-116; doi 10.1097/PGP.0b013e3181f4de77
[12] Muminhodzic L., Bogdanovic G., Ultrasonographic signs of
partial hydatidiform mole, Med Arch, 2013, 67, 205-208
[1] imercati A., de Gennaro A.C., Cobuzzi I., Grasso S., Abruzzese
[13] Steller M.A., Genest D.R., Bernstein M.R., Lage J.M., Goldstein
M., Fascilla F.D., et al., Two cases of complete hydatidiform
D.P., Berkowitz R.S., Natural history of twin pregnancy with
mole and coexistent live fetus, J Prenat Med, 2013, 7, 1-4
complete hydatidiform mole and coexisting fetus, Obstet
[2] Candelier J.J., The hydatidiform mole, Cell Adh Migr, 2016, 10,
Gynecol, 1994, 83, 35-42
226-235; doi 10.1080/19336918.2015.1093275
[14] Jones W.B., Lauersen N.H., Hydatidiform mole with coexistent
[3] Rohilla M., Singh P., Kaur J., Jain V., Gupta N., Prasad G.R.,
fetus, Am J Obstet Gynecol, 1975, 122, 267-272
Individualistic approach to the management of complete
[15] Peng H.H., Huang K.G., Chueh H.Y., Adlan A.S., Chang S.D.,
hydatidiform mole with coexisting live fetus, Eur J Obstet
Lee C.L., Term delivery of a complete hydatidiform mole with
Gynecol Reprod Biol, 2015, 191, 39-42; doi 10.1016/j.
a coexisting living fetus followed by successful treatment
ejogrb.2015.05.017
of maternal metastatic gestational trophoblastic disease,
[4] Gupta K., Venkatesan B., Kumaresan M., Chandra T., Early
Taiwan J Obstet Gynecol, 2014, 53, 397-400; doi 10.1016/j.
Detection by Ultrasound of Partial Hydatidiform Mole With a
tjog.2013.02.005
Coexistent Live Fetus, WMJ, 2015, 114, 208-211; quiz 212
[16] Sebire N.J., Prenatal diagnosis and management of twin
[5] Wee L., Jauniaux E., Prenatal diagnosis and management
pregnancies complicated by a co-existing molar pregnancy,
of twin pregnancies complicated by a co-existing molar
Prenat Diagn, 2006, 26, 373; doi 10.1002/pd.1380
pregnancy, Prenat Diagn, 2005, 25, 772-776; doi 10.1002/
[17] Frederick L., Greene, Carolyn C., Gestational tropho-blastic
pd.1272
tumors, Ajcc Cancer Staging Handbook, 2002, 8, 323-328
[6] Chu W., Chapman J., Persons D.L., Fan F., Twin pregnancy
[18] Dickinson J.E., Harcourt E., Murch A., The selective use of
with partial hydatidiform mole and coexistent fetus,
rapid aneuploidy screening in prenatal diagnosis, Aust N
Arch Pathol Lab Med, 2004, 128, 1305-1306; doi
Z J Obstet Gynaecol, 2009, 49, 28-33; doi 10.1111/j.1479-
10.1043/1543-2165(2004)128<1305:TPWPHM>2.0.CO;2
828X.2008.00939.x
[7] Ingec M., Borekci B., Altas S., Kadanali S., Twin pregnancy
[19] Wang Y., Qian H., Wang J., Medical termination of a partial
with partial hydatidiform mole and coexistent normal
hydatidiform mole and coexisting fetus during the second
fetus, J Obstet Gynaecol, 2006, 26, 379-380; doi
trimester: A case report, Oncol Lett, 2015, 10, 3625-3628; doi
10.1080/01443610600618747
10.3892/ol.2015.3743