Case Report

Suggested Method for the Management of Heterotopic Cervical

Pregnancy Leading to Term Delivery of the Intrauterine Pregnancy:
Case Report and Literature Review
Elias Tsakos, MD, Nikolaos Tsagias, MD, PhD*, and Konstantinos Dafopoulos, MD, PhD
From EmbryoClinic, Thessaloniki, Greece (Dr Tsakos), Department of Obstetrics and Gynecology, Democritus University of Thrace, Xanthi, Greece (Dr
Tsagias), and Department of Obstetrics and Gynecology, Faculty of Medicine, School of Health Sciences, University of Thessaly, Larissa, Greece (Dr
Dafopoulos).

ABSTRACT This is a case report and literature review regarding early diagnosis and management of a cervical heterotopic pregnancy. A
41-year-old gravida 2 para 0 with premature ovarian failure was treated successfully in an in vitro fertilization program with
donor oocytes. A transvaginal ultrasound scan revealed the presence of a heterotopic pregnancy, with an intrauterine embryo
and an intracervical embryo. Both embryos had positive heartbeats. Aspiration of the cervical pregnancy was followed by
Foley catheter placement and cervical cerclage suturing. Monitoring of the patient resulted in elective uncomplicated cesarean
section delivery at 38 weeks following an uneventful pregnancy. Given the lack of guidelines for the management of hetero-
topic cervical pregnancy, we reviewed the literature suggesting the most effective method. The value of early diagnosis
and management is concluded from the literature, in support of our management principles. Journal of Minimally Invasive
Gynecology (2015) -, -–- Ó 2015 AAGL. All rights reserved.
Keywords: Donor oocyte; Foley catheter; Heterotopic cervical pregnancy; In vitro fertilization; Shirodkar cervical cerclage

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Successful management of cervical heterotopic
pregnancy remains a challenge, given the lack of current
guidelines. The guiding management principle remains the
safe and effective removal of the cervical pregnancy while
protecting the intrauterine pregnancy (IUP) and preserving
fertility.
Cervical pregnancy is a very rare form of ectopic
pregnancy, with a reported incidence ranging between 1 in
1000 to 1 in 18 000 of all pregnancies [1,2] and 1% of all
ectopic pregnancies [3]. Cervical implantation is associated
with previous cesarean section delivery, dilation and
Disclosure: None declared.
Corresponding author: Nikolaos Tsagias, MD, PhD, Department of
Obstetrics and Gynecology, Democritus University of Thrace, Papaflessa
8, Alexandropoulis 57500, Greece.
E-mail: tsagiasnikos@yahoo.gr
Submitted February 9, 2015. Accepted for publication March 12, 2015.
Available at www.sciencedirect.com and www.jmig.org
1553-4650/$ - see front matter Ó 2015 AAGL. All rights reserved.
http://dx.doi.org/10.1016/j.jmig.2015.03.009
curettage procedures, and in vitro fertilization (IVF)
techniques [4].
Heterotopic pregnancy is an extremely rare condition,
with the cervix the rarest site and the fallopian tube the
most common [5]. Early diagnosis of a heterotopic preg-
nancy is often difficult owing to its asymptomatic nature in
initial stages. A high degree of clinical suspicion is required
to avoid life- and fertility-threatening complications associ-
ated with late presentation.
Despite improvements in ultrasound technology, the risks
of maternal morbidity and mortality and fertility compromise
associated with cervical heterotopic pregnancy remain. Early
diagnosis is crucial to avoid complications and successfully
apply conservative methods aimed at preserving the coexist-
ing intrauterine pregnancy. Several methods have been
described in the literature for the management of cervical het-
erotopic pregnancies, including aspiration, cervical curettage,
forceps extraction, cervical cerclage, Foley catheter place-
ment, and local injection of potassium chloride (KCl) or
2 Journal of Minimally Invasive Gynecology, Vol -, No -, -/- 2015
Fig. 1
Heterotopic cervical pregnancy and IUP.
methotrexate. Various combinations of the foregoing methods
have been described as well. Owing to the rarity of heterotop-
ic cervical pregnancy and the variety in clinical presentation,
no guidelines exist for its management.
Here we present a case study of management of a hetero-
topic cervical pregnancy. We also present the results of a
literature review comparing our novel method with previ-
ously described techniques.
Case Report
An asymptomatic 41-year-old gravida 2 para 0 presented
at 5 weeks of pregnancy following IVF treatment with the
use of donor oocytes owing to premature ovarian failure.
In the previous year, the patient experienced an ectopic cer-
vical pregnancy after IVF, which was treated with dilation
and curettage. She had no other history of cervical and/or
uterine surgery. She had an unremarkable medical and surgi-
cal history, and transvaginal ultrasound scan and hysterosal-
pingography with the use of radiopaque dye performed
immediately before IVF were normal.
Fig. 2
(A) Color Doppler transvaginal ultrasound image showing the cervical pregnancy with peritrophoblastic flow. (B) Viable cervical pregnancy with fetal
heartbeat.
The patient had undergone embryo transfer of 2
blastocyst-stage embryos. Her serum b-human chorionic
gonadotropin (b-HCG) level at 9 days after embryo transfer
was 172 mIU/mL, and 2 days later increased to 417.4 mIU/
mL. At 5 weeks, 3 days of gestation, a transvaginal ultra-
sound examination (GE Logic P6 ultrasound machine with
an E8CS probe, 4–10 MHz; GE Healthcare, Little Chalfont,
UK), revealed a heterotopic IUP and cervical pregnancy. The
2 gestational sacs were of equal size at 11.2 mm, with yolk
sacs present.
The patient and her husband, both medical doctors, were
counseled regarding the diagnosis, possible sequelae, and
management options. They agreed with our suggestion for
expectant management in the first instance. An emergency
mobile number with 24-hour medical coverage was provided
to the patient, and open access to the hospital was arranged.
The patient was asymptomatic, and transvaginal ultrasound
examination 1 week later revealed the following findings: in-
trauterine and cervical pregnancy, both singleton (Fig. 1).
The cervical pregnancy demonstrated a peritrophoblastic
blood flow with a normal fetal heart rate and compatible
with dates (i.e., 6 weeks and 3 days), detected by transvagi-
nal color Doppler examination (Fig. 2). The patient was
counseled about treatment options, lack of management
guidelines, and possible risks associated with conservative
methods, namely miscarriage, hemorrhage, blood transfu-
sion, preterm delivery, and hysterectomy. The patient con-
sented to conservative management aiming to conserve the
IUP. Four units of whole blood and 4 units of fresh-frozen
plasma were cross-matched and a general anesthetic was
administered.
With the patient in lithotomy position, aseptic conditions
were applied, and a Foley catheter was inserted into the
bladder. The catheter was clamped to allow bladder filling
through intravenous fluids (1 L of Ringer’s lactate; Baxter,
Deerfield, IL) for facilitation of transabdominal ultrasound
imaging. The cervix showed signs of insufficiency: in mid-
position and soft, with a cervical canal length of 2.5 cm
Tsakos et al. Management of Heterotopic Cervical Pregnancy
and 1 cm dilation of the external cervical os. The assistant
held the transabdominal ultrasound probe (GE Logic P6,
1.5–3.5 MHz) as the operator inserted a silastic no 6. suction
curette (single-use flexible curette; MedGyn Products, Addi-
son, IL) without previous cervical dilation. The curette was
slowly inserted 1 cm into the canal and positioned 2–3 mm
below the cervical sac, as demonstrated by transabdominal
ultrasound. The curette was connected to a suction pump
(Medela, Baar, Switzerland), mild suction slowly increased
to 400 mmHg was applied, and the cervical sac was slowly
suctioned; this was clearly demonstrated on ultrasound scan.
On completion of suction, the pump was turned off, and the
curette was slowly removed. Moderate hemorrhage
commenced immediately after removal, and a Foley catheter
no. 18 was inserted into the cervical canal at the level of just
below the internal os and 2 cm from the external os. The Fo-
ley catheter was inflated with 4 mL of normal saline solution,
which instantly controlled the cervical bleeding. A Shirod-
kar cerclage Mercilene tape suture (5 mm wide; Ethicon,
Somerville, NJ) was applied around the cervix below to
the internal os, higher from the Foley catheter, owing to
the clinically obvious cervical insufficiency, for hemostatic
purposes and to secure the Foley catheter placement. An in-
traoperative transvaginal ultrasound examination confirmed
the viability of the intrauterine pregnancy, correct placement
of the cervical Foley catheter and Shirodkar suture, and the
absence of a hematoma. The bladder Foley catheter was un-
clamped at the end of the procedure.
Intraoperative treatment included antibiotic prophylaxis
with intravenous amoxicillin 875 mg plus clavulanic acid
125 mg and subcutaneous low molecular weight heparin
4000 IU. Low molecular weight heparin was continued post-
operatively and for the entire antenatal period due to high
thrombosis risks associated with IVF pregnancy, maternal
age and reduced maternal physical activity.
On the first postoperative day, the patient was comfort-
able and hemodynamically stable, with a hemoglobin con-
centration of 11.3 g/dL, minimal brown vaginal spotting,
and normal transvaginal ultrasound findings. A total of 1
mL of normal saline was removed from the cervical Foley
catheter. On the second postoperative day, she was asymp-
tomatic with normal ultrasound findings. Another 1 mL of
normal saline was removed from the Foley catheter in the
morning, and the catheter was removed at noon. The patient
was discharged home late afternoon on the second postoper-
ative day with instructions to avoid any physical activity in
the first week and with clear instructions in case of emer-
gency.
Histopathological examination of the cervical pregnancy
revealed the presence of chorionic villi and fetal products of
conception. There was no evidence of any abnormality.
The patient was closely monitored throughout pregnancy
and had an unremarkable antenatal period. The IUP pro-
gressed normally, and the cervix remained intact with the
Shirodkar suture in situ until 38 weeks gestation (Fig. 3).
At 38 weeks, an uncomplicated cesarean section was per-
3
Fig. 3
Full-term pregnancy with an intact, 3.64-cm-long cervix. Shirodkar
cerclage sutures (arrow) in the cervix protecting the IUP.
formed, and a healthy female infant weighing 2900 g was
delivered. The Shirodkar suture was removed uneventfully
after birth. The local Institutional Review Board exempted
our case from the need for approval.
Discussion
To our knowledge, this is the 44th case report of hetero-
topic cervical pregnancy in the world literature (based on a
search of the Embase, Pubmed, and Scope databases), and
the first case in which aspiration suction of the cervical preg-
nancy was followed by cervical Foley catheter placement
and Shirodkar cerclage, leading to an uneventful full-term
delivery. Similar successful management was reported by
Moragianni et al [6], who used a Foley catheter fixed by
cerclage sutures for 48 hours in an effort to control the cer-
vical bleeding. The use of a Foley catheter and cerclage su-
tures was also reported by Hafner et al [7]; however, in that
case the IUP was terminated with the use of methotrexate ac-
cording to the patient’s desire.
Our literature review revealed no typical method or
guidelines for the management of heterotopic pregnancy.
Reported management depends on the time and mode of pre-
sentation in each case (e.g., early or delayed, asymptomatic
or acute), the patient’s wish to preserve the intrauterine preg-
nancy, hospital setting, and the physician’s skills. The man-
agement of cervical heterotopic pregnancy with Foley
catheter placement also has been described in 2 other cases,
both of which had a successful full-term outcome without
complications [8,9]. It is known that the inflated Foley
catheter may be used successfully for cervical bleeding
control associated with cervical ectopic pregnancies [10–
12]; this method has been reported to achieve hemostasis
in 92.3% of cases [7].
Various methods of cervical suturing have been described
in the literature. Cervical cerclage suturing was initially
4 Journal of Minimally Invasive Gynecology, Vol -, No -, -/- 2015

Table 1
Reported heterotopic pregnancies managed with cervical sutures, Foley catheter placement, or a combination of the 2 methods

Reference Treatment Pregnancy outcome

Present case Foley catheter 1 Shirodkar cerclage after aspiration FT delivery (38 w; CS)
Moragianni et al [6] Foley catheter 1 cerclage sutures for 48 h after forceps extraction FT delivery (39 w; CS)
Uysal et al [9] Foley catheter 1 cervical cerclage for 48 h after KCl injection and aspiration FT delivery (38 w)
Hafner et al [7] Foley catheter 1 cerclage 1 ligation of the descending cervical branches Termination (patient’s wish)
of the uterine arteries 1 methotrexate
Mashiach et al [16] Shirodkar cerclage after aspiration FT delivery (39 w; VD)
Faschingbauer et al [17] Shirodkar cerclage after curettage FT delivery (39 w 1 3 d; VD)
Jozwiak et al [15] McDonald cerclage suture FT delivery (38 w; CS)
Chen et al [14] Cerclage sutures after aspiration and KCl injection FT delivery (38 w; CS)
Davies et al [13] Digital evacuation and cervical cerclage Miscarriage
Kim et al [8] Foley catheter FT delivery (37 w; CS)

CS 5 cesarean section; FT 5 full term; VD 5 vaginal delivery.

reported by Davies et al [13] in a case of heterotopic preg-
nancy. Cervical stay sutures were used after aspiration by
Chen et al [14], and a modified method of leaving cervical
sutures in situ for 48 hours, ensuring fixation and stability
of the endocervical Foley catheter, was described by Mora-
gianni et al [6]. Cerclage suturing followed by bilateral liga-
tion of the descending cervical uterine artery branches was
reported by Hafner et al [7]. A McDonald cerclage technique
was also described for managing a cervical heterotopic preg-
nancy [15].
To our knowledge, Shirodkar cerclage suturing as an
elective method for the management of cervical pregnan-
cies, both ectopic and heterotopic, was first described by Ma-
shiach et al [16]. Based on its successful application in 4
cases, the authors suggested that Shirodkar cerclage may
be the treatment of choice for heterotopic cervical preg-
nancy, More recently, Fauschingbauer et al [17] reported
Shirodkar cerclage suturing following curettage of a cervical
pregnancy that finally ended in a term delivery. It is
extremely significant that all but the first cases resulted in
full-term delivery (Table 1).
Medical treatments associated with successful outcome
also have been described with the use of KCl in most cases;
this was associated with various complications. Metho-
trexate use also has been reported in some studies, and in a
few studies hypersmolar glucose was applied. To date, in
13 out of 14 heterotopic pregnancies treated with KCl, the
IUP was preserved. In 1 study, there was no follow-up after
the 12th week of gestation [18]. However, in the majority of
theses cases, major antenatal and intrapartum hemorrhage

Table 2
Reported heterotopic cervical pregnancies managed with KCl injection

Author Treatment Pregnancy outcome Complications

Majumdar et al [22]
Gyamfi et al [19]
Olah [20]
Monteagudo et al [23]
Carreno et al [24]
Kumar et al [25]
Deka et al [21]
Al-Azemi et al [26]
Frates et al [27]
Wu et al [28]
Honey et al [21]
Proporic et al [18]
KCl injection PT delivery (31 w; CS) Bleeding
KCl injection PT delivery due to vaginal bleeding (31 w; CS) Bleeding; hysterectomy, transfusion
KCl injection PT delivery due to vaginal bleeding (36 w; CS) Bleeding; hysterectomy
KCl injection PT delivery (34 w; CS) Bleeding; placenta previa accreta from
the cervical placenta, left in situ
KCl injection PT delivery (36 w; VD) No complications
KCl injection PT delivery (35 w; CS) due to preeclampsia Bleeding; ligation of both internal iliac
arteries, transfusion
KCl, MTX injection PT delivery (36 w1 4 d; CS) due to preeclampsia
KCl, MTX injection PT delivery (30 w; /S) due to premature
rupture of membranes
KCl injection PT delivery (35 w; CS)
KCl injection Delivery
KCl injection Miscarriage of the IUP 1 w after KCl injection Bleeding; transfusion, hysterectomy
KCl injection Follow-up to week 12

CS 5 cesarean section; MTX 5 methotrexate; PT 5 preterm; VD 5 vaginal delivery.

The cases reported by Uysal et al [9] and Chen al. [14] are included in Table 1 owing to additional catheter placement and cerclage sutures, respectively.
Tsakos et al. Management of Heterotopic Cervical Pregnancy
were reported. Bleeding as a complication was reported in 6
cases, and in 3 cases a cesarean hysterectomy was performed
[19–21]. It is important to emphasize that the majority of
cases managed with KCl resulted in preterm delivery,
owing mainly to severe antepartum hemorrhage (Table 2).
Only 2 cases treated with KCl resulted in term delivery
[9,14]; in those cases, an intracervical Foley catheter and a
cervical cerclage were placed respectively (Table 1). In 2
other cases, the pregnancy was interrupted at 36 weeks, 4
days gestation and 35 weeks gestation, respectively, owing
to preeclampsia [25,29].
Methotrexate administration also has been reported in
some cases of cervical pregnancy; in the majority of these
cases, the IUP was terminated. Only 3 cases of successful
preservation of the IUP associated with methotrexate injec-
tion have been reported to date [26,29,30]. Two studies
reported that the use of hypersmolar glucose was
associated with preservation of the IUP. One of these was
complicated by a massive postpartum hemorrhage [31],
and the other resulted in preterm twin delivery [32]. Other
attempts to manage cervical pregnancy, including hystero-
scopic ablation and angiographic embolization, have been
reported as well [15,21].
It is now widely accepted that certain management op-
tions suitable for cervical ectopic pregnancy (e.g., metho-
trexate administration) may be contraindicated in cases of
heterotopic cervical pregnancy in which there is a wish to
maintain the IUP. However, no ideal method for the manage-
ment of heterotopic cervical pregnancy exists in the litera-
ture, and there are no recommendations and guidelines for
safe and effective treatment.
Conclusion
This literature review and comparison of the main exist-
ing management methods suggests that cerclage cervical su-
turing and intracervical Foley catheter placement are the
safest techniques associated with term delivery. In our
case, using this method, the cervical pregnancy was removed
while the IUP was preserved, resulting in a term delivery.
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