Vol.2, No.9, 568-572 (2013) Case Reports in Clinical Medicine
http://dx.doi.org/10.4236/crcm.2013.29143
Selective reduction of a triplet heterotopic cervical
pregnancy after embryo transfer
Delphine Delrieu1,2*, Roland Antaki1,2, Simon Phillips1, Isaac-Jacques Kadoch1,2
1OVO Clinic, Montreal, Canada;
*Corresponding Author:
2University of Montreal, Department of Obstetrics and Gynaecology, Saint-Luc Hospital (CHUM), Montreal, Canada
Received 16 August 2013; revised 14 September 2013; accepted 10 October 2013
Copyright © 2013 Delphine Delrieu et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. In accor-
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ABSTRACT
Cervical pregnancy is rare and heterotopic cer-
vical pregnancy is more uncommon ever. As-
sisted Reproduction Technolog y enhances these
pregnancies. The diagnosis and the treatment
which are in emergency can be sometimes dif-
ficult. This article is a case report of a triplet
heterotopic cervical pregnancy after embryo
transfer, and a scientist review of the literature
found in MEDLINE. To date, in the English lit-
erature, we found only 7 cases report of triplet
heterotopic cervi cal pregnancies.
Keywords: Heterotopic Pregnancy; Cervical
Pregnancy; Ectopic Pregnancy; IVF; Emb ry o
Transfer; Assisted Reproduction Technology
1. INTRODUCTION
Heterotopic pregnancy is an association between an
intrauterine and an ectopic pregnancy (tubal or more
rarely ovarian, cervical, abdominal). The first description
comes from Duvernay in 1761 during an autopsy [1]. It
is a rare natural phenomenon (less than 1/30000 preg-
nancies) [2,3], but its rate increases with the introduction
of Assisted Reproductive Technology (ART) (0.8% - 3%
after IVF) [4,5].
Cervical pregnancies are very rare (1/2500 - 18000
pregnancy and <1% ectopic pregnancies) [6-9] and het-
erotopic cervical pregnancies, usually resulting from
ART, are even more rare [10].
This is the first report of an ongoing pregnancy after a
selective aspiration of the cardiac fluid of a triplet het-
erotopic cervical pregnancy.
2. CASE REPORT
Our patient was a 36 years old, with 3 previous spon-
taneous conceptions and deliveries, with an another
partner. She underwent a tubal ligation after her last de-
livery. A few years later, she consulted with a new part-
ner. Her partner had a normal sperm count. So she un-
derwent a tubal reanastomosis which was unsuccessful
with a bilateral tubal obstruction seen on her post opera-
tive hysterosalpingography. After discussing the alterna-
tives, the couple decided to undergo IVF.
The patient received a long agonist protocol using a
gonadotrophin-releasing hormone (GnRH) agonist (Su-
prefact; Sanofi-Aventis) introduced in the luteal phase of
the previous cycle at a dose of 0.5 mg SC daily and then
decreased to 0.25 mg daily at the initiation of gonad-
otrophin stimulation. The patient received 225 IU daily
of hMG (MENOPUR; Ferring, Canada) that was in-
creased to 300 IU in association with 150 IU of recom-
binant FSH (GONAL-F; EMD Serono Canada). Final
oocyte maturation was triggered by 10000 IU human
chorionic gonadotrophin (Pregnyl, Merck, Canada). The
patient underwent oocyte retrieval 36 hours after receiv-
ing hCG; 14 oocytes were retrieved. After insemination
with sperm, we obtained 9 embryos. The patient had an
ultrasound guided embryo transfer of 3 day3 embryos
(8C2-3, 8C2-3 and 10C2). The transfer technique was
described as easy by the physician. Two weeks later,
hCG level was 186.8 UI/L and 331.5 UI/L two days after.
At 8 weeks and 3 days of amenorrhea, a trichorionic
triamniotic triplet pregnancy with one gestationnal sac in
the cervix was discovered at endovaginal ultrasound. The
3 embryos had a positive heart beat.
After discussing the situation with the couple, they de-
cided to undergo a embryonic reduction of the cervical

D. Delrieu et al. / Case Reports in Clinical Medicine 2 (2013) 568-572 569
pregnancy. They understood and accepted the procedure,
including the risk of bleeding and the risk of losing the
three embryos.
At 8 weeks and 4 days of amenorrhea, in the operating
room, the patient had para cervical block with XYLO-
CAÏNE 1%. We used the 7.5 MHZ endovaginal ultra-
sound probe (Voluson E8, GE, Healthcare, Baie d’Urfé,
QC, Canada) with a needle guide. Using an egg collec-
tion needle (OPS 30cm, 19G, CCD, France), we aspi-
rated the amniotic fluid and then the cardiac blood of the
cervical embryo. No heart beat was seen for the cervical
embryo at the end of the procedure, and the intrauterine
embryos had normal heart beat. The procedure was easy
and the patient did not have any pain or bleeding.
A 12 weeks ultrasound showed a dichorionic diamni-
otic ongoing twin pregnancy
The patient had a normal pregnancy that was compli-
cated by premature labor, an emergency C-section was
performed at 25 weeks; she delivered a girl (660 g) and a
boy (620 g).
3. DISCUSSION
Heterotopic cervical pregnany is a very rare event.
ART use has increased the rate of heterotopic pregnan-
cies with unusual localisation like cervix or cesaerian
section scar [11,12].
The diagnosis of a cervical pregnancy can be difficult
and delayed [13-15]. The main symptoms are bleeding
during the first trimester, abnormal hCG increase. The
transvaginal ultrasound is usually diagnostic (the first
report has been described by Raskin in 1978) [16] but
magnetic resonance imaging (MRI) has been necessary
in some cases to make the definitive diagnosis [17,18].
The diagnosis is confirmed by finding an empty uterine
cavity, in association with a gestationnal sac with an em-
bryo (with a positive or negative heart beat) in the endo-
cervical canal below the internal os level. A heterotopic
cervical pregnancy can be even more difficult to diag-
nose, because someone can focus on the intrauterine
pregnancy without seeing the cervical one.
Cervical pregnancy is a therapeutic emergency be-
cause of the risk of severe hemorrhage. There were even
recent reports of death following a heterotopic cervical
pregnancy [19].
Different treatments for ectopic cervical pregnancy are
described in the literature: systemic Methotrexate injec-
tion [20-22], uterine artery embolization or ligation [23]
[24], transvaginal ultrasound-guided aspiration [23], KCl
solution [25], Methotrexate [13,26,27] or hyperosmolar
glucose injection into the gestational sac [28], cervical
curettage with balloon tamponnade [29-31] or Shirodkar
cerclage to make hemostasis [32]. Olav describes a hys-
teroscopy resection of a cervical pregnancy because of
bleeding after Methotrexate treatment [33].
There is no consensus on the technique to use for
treatment. Prognostic factors that affects the effective-
ness of the conservative treatment are especially the age
of pregnancy [34,35]. Sometimes, hysterectomy is inevi-
table because of severe bleeding [13,36,37].
In our case, systemic Methotrexate and uterine artery
embolization or ligation were not an option because of
the presence of an ongoing intrauterine pregnancy.
We chose the selective reduction by the fetal heart as-
piration technique, but Sijanovic et al. tried selective
Methotrexate injection, and had a good result [26]. Some
articles describe the injection of KCl in fetal heart or
gestationnal sac [19,38-46]. Some authors injected Ac-
tinomycin D [47]. Jozwiak et al. resected the cervical
pregnancy by hysteroscopy and coagulated the bleeding
sites with roller-ball. The remaining intra uterine preg-
nancy then continued successfully to term [48]. Kim et al.
and Vasiliki et al. aspirated the cervical pregnancy and
achieved hemostasis by compression with pediatric
Foley catheter [30,49].
All these techniques have three goals: minimize the
bleeding, preserve the fertility and the intrauterine preg-
nancy. We have to keep in mind that if these conservative
treatments fail or if the patient starts to bleed severely,
there would no choice but to sacrifice the intrauterine
pregnancy with the first treatments methods described.
The mechanism to explain a cervical pregnancy is un-
clear. Cervical or uterine surgeries in the past (curettage
or cesarean), seem to enhance these ectopic pregnancies
[36,37,50,51]. Does the problem come from an unrecep-
tive endometrium?
For the rare spontaneous cervical pregnancies, some
authors wonder if the transport of the ovum would be too
quick and that the embryo arrives in the uterine cavity
with an unprepared endometrium [52,53].
A chromosomal abnormality in the embryo is another
hypothesis [51,54].
As mentioned before, these ectopic pregnancies are
more common after ART and specially after IVF [40,48,
55-58]. Furthermore, in the english literature, we found
only 7 cases report of triplet heterotopic cervical preg-
nancies (6 with intrauterine twins and one with cervical
twins) [5,28,44,59-62]. None of them were spontaneous,
all these pregnancies were the result of embryo transfer
after IVF. So we can say that IVF and transferring more
than one embryo are risk factors for heterotopic preg-
nancies.
4. CONCLUSION
Historically, many women with a cervical ectopic or
heterotopic cervical pregnancy underwent hysterectomy.
With the development of transvaginal ultrasound, the
diagnosis of these rare pregnancies has become easier
and quicker. A conservative method should be tried first,

D. Delrieu et al. / Case Reports in Clinical Medicine 2 (2013) 568-572
570
especially as these pregnancies are in infertile patients
undergoing ART. This is the first report of an ongoing
pregnancy after a selective aspiration of the cardiac fluid
of a triplet heterotopic cervical pregnancy.
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