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CASE REPORT www.ivflite.org

Heterotopic cer vical pregnancy:

Conservative treatment with transvaginal
Quick Response Code
embryo aspiration
Mete Isikoglu
Gelecek, The Center For Human Reproduction, Antalya, Turkey
Address for Correspondence: Dr. Mete Isikoglu, Gelecek, The Center For Human Reproduction, Antalya,
Turkey E‑mail: misikoglu@hotmail.com

About the Author

Mete Isikoglu, M.D. is consultant IVF practitioner in Gelecek-The Center For Human Reproduction, Antalya,
Turkey. He has been part of the scientific committee of the World Association of Reproductive Medicine (WARM)
and he is a member of the Antalya Chamber of Medicine, Turkish Society of Gynecology and Obstetrics, ESHRE
and the Preimplantation Genetic Diagnosis International Society (PGDIS). He has many international and national
publications and presentations. He has also participated at several international conferences and contributed nearly
15 textbook chapters. He is a peer reviewer for Fertility Sterility, European Journal of Obstetrics Gynecology and
Reproductive Biology, Reproductive Biomedicine Online, Journal of Assisted Reproduction and Genetics, International Journal of Fertility
and Sterility, IVF Lite and Turkish Journal of Obstetrics and Gynecology. His field of interest is Gynecologic Endoscopic Surgery and
holds the Bachelor in Endoscopy certificate awarded by the European Academy of Gynecological Surgery.

AB STR A C T
To discuss two cases of heterotopic cervical pregnancy  (HCP) managed with transvaginal embryo reduction. Two patients
diagnosed with HCP after assisted reproductive treatment were treated with ultrasonographically guided transvaginal embryo
aspiration of ectopic fetus. In one case, the pregnancy resulted in healthy delivery while the second case was terminated due to
chorioamnionitis followed by preterm premature rupture of membranes at 23 weeks of gestation. This minimally invasive approach
in an hemodynamically stable patient can be considered in the management of a first‑trimester HCP. However, the patient must
be informed for the potential risks related to the procedure.
Key Words: Assisted reproductive technology, cervical pregnancy, heterotopic cervical pregnancy, selective embryo
reduction, selective fetal reduction

INTRODUCTION early pregnancy, more cases of cervical ectopic pregnancy

Cervical pregnancy is a life‑threatening form of ectopic
pregnancy that implants within the endocervical canal.
It accounts for <0.15% of all ectopic pregnancies.[1‑3]
Heterotopic pregnancy, defined as the coexistence of an
intrauterine pregnancy (IUP) and an ectopic pregnancy
is a rare entity with an incidence of 1 in 10.000–50.000
in natural gestations. With the extended use of assisted
reproductive technology (ART), the incidence rose nearly
70 fold.[4,5] Heterotopic cervical pregnancy (HCP) is even
more unusual. With the increase in use of ART worldwide
and more liberal use of transvaginal sonography during
IVF Lite Foundation Published by Medknow. All rights reserved.
Please cite this article as: Isikoglu M. Heterotopic cervical pregnancy: Conservative treatment with transvaginal embryo aspiration. IVF Lite 2015;2:37-41.
DOI: 10.4103/2348-2907.158745
are being reported.
So far, successful management resulting in healthy live birth
of the IUP was reported in a few cases of HCP.[6]
The low incidence of the entity and improving treatment
options necessitate this rare condition to be discussed
repeatedly. We present two patients with HCP for whom
a conservative treatment approach was applied. Treatment
options are also revised. Approval of the Institutional
Review Board was obtained prior to submitting the
manuscript.

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Isikoglu: Treatment of heterotopic cervical pregnancy
CASE REPORTS
Case 1
A couple presented to our center with an infertility history
of 8 years. The lady (40 years) had migraine in her past
medical history. She had no treatments for infertility and
no operations in surgical history.
After standard investigation work‑up, the couple was
enrolled in ART program with the diagnosis of “mild male
factor infertility.” The treatment was performed according
to short antagonist protocol. A total of 7 metaphase II
oocytes were retrieved and after intracytoplasmic sperm
injection, two embryos were transferred on day 2. The
result of blood human chorionic gonadotropin (hCG) assay
on 14th day after embryo transfer (ET) was 680 IU/mL.
Twenty‑one days after ET (in 5 + 2/7th gestational week),
transvaginal ultrasound demonstrated a gestational sac in
uterine cavity, repeat exam at 7th week showed a second
gestational sac located in the cervical canal and live
embryos in both sacs with crown‑rump lengths (CRLs)
corresponding to gestational age [Figure 1]. The patient was
asymptomatic. Selective embryo reduction was planned for
the following day. Elimination of the cervical gestation was
performed under sedation anesthesia with propofol. After
preparing the perineum and vagina with poviod‑iodine and
sterile draping in litothomi position, an ultrasonographically
guided 17 gauge‑follicle aspiration needle (MDT® Medical
Instrument Division BV, The Netherlands) which was
attached to a 20 mL syringe was inserted through the vagina
and the embryo was aspirated by creating negative pressure
via pulling the plunger. Negative pressure was maintained
until complete absence of the embryo was observed.
Single dose 1 g cephazoline was administered intravenous
Figure 1: Preoperative sonographic image of heterotopic cervical
pregnancy. Upper arrow: Fundal gestational sac and embryo; Lower
arrow: cervical gestational sac and embryo
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peroperatively. After 3 h of bed rest, live intrauterine
embryo and terminated cervical gestation were rechecked
by ultrasound and the patient was discharged.
Sonographic examination 1 week later revealed live
intrauterine fetus with appropriate CRL and an intracervical
remnant of the cervical sac. Serial ultrasound examinations
revealed that the intrauterine fetus showed appropriate
growth and that the intracervical fluid appearance was
still existing at 15th weeks of gestation measuring nearly
5 cm in longer dimension [Figure 2]. This fluid collection
got smaller gradually and disappeared by 22 weeks of
gestation. With regular antenatal follow‑up, the pregnancy
resulted in healthy delivery at 39 + 2/7 weeks of gestation
by cesarean section due to cephalopelvic disproportion and
a live 3350 g female baby was born.
Case 2
This patient was referred to our center for ART with
the diagnosis of “bilateral tubal occlusion.” Duration of
infertility was 1 year. The lady (35 years) had open surgery
for appendicitis and she had no treatments for infertility
before. The treatment was performed according to short
antagonist protocol. A total of 5 oocytes were obtained and
after in vitro fertilization, two embryos were transferred on
day 2. ET procedures in both cases were done as described
elsewhere.[7] The result of blood hCG assay on 12th day
after ET was 347 IU/mL.
Eighteen days after ET (in 4 + 4/7th gestational week),
transvaginal ultrasound demonstrated a gestational sac in
the uterine cavity, and a smaller sac in the cervical canal.
At 5 + 3/7 gestational week, the patient presented with
spotting like mild vaginal bleeding. Sonography showed
live embryo in the intrauterine sac with a fetal heart rate
of 100/min and fluid collection in the cervical canal.
Follow‑up sonography one week later showed live embryos
both in the intrauterine and the cervical sacs with CRLs
corresponding to gestational age [Figure 3]. Elimination
of the cervical gestation was performed as described in
the previous case. The reduction procedure itself took
a b
Figure 2: Sonographic image at 15th weeks of gestation. Intracervical
fluid collection still exists. (a) Measurement of the cervical canal.
(b) Measurement of the fluid collection
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Isikoglu: Treatment of heterotopic cervical pregnancy
Figure 3: Preoperative sonographic image of heterotopic cervical
pregnancy. Upper arrow: Fundal gestational sac and embryo; Lower
arrow: cervical gestational sac and embryo
1 min. Few minutes after the completion of the reduction,
severe bleeding commenced. Since persistent cervical
tamponade with sponges failed to stop the bleeding,
McDonald cervical cerclage suture was applied. The
bleeding stopped completely just after the cerclage. The
patient was hospitalized for 6 h and discharged after live
intrauterine embryo and terminated cervical gestation
was rechecked by ultrasound. Follow‑up sonographic
examination 1 week later revealed live intrauterine fetus
with appropriate CRL and a heterogenous intracervical
remnant of the cervical sac measuring 1.1 cm [Figure 4].
The cervical remnant disappeared by 14 weeks of gestation.
The patient experienced painless pinky vaginal discharge
without any contraction at 18th gestational week. White
blood cells and C‑reactive protein results were normal.
Thereafter, while the antenatal follow‑up was continuing
elsewhere, she was admitted to the emergency department
with the complaints of odorous vaginal discharge and
crampy groin pain at 23 weeks of gestation. She was
diagnosed with chorioamnionitis followed by preterm
premature rupture of membranes, and the pregnancy was
terminated under combined antibiotherapy. The baby did
not survive, and the lady recovered without any problem
and was discharged 3 days after delivery.
DISCUSSION
Heterotopic cervical pregnancy is an extremely rare
phenomenon, and most cases are related to ART. A recent
review has identified only 37 cases reported in the English
language literature all but four of which are the result of
ARTs.[6]
Although cervical component of HCP has the potential for
high morbidity due to massive hemorrhage; the mortality
IVF Lite | January-April 2015 | Vol 2 | Issue 1
Figure 4: Sonographic image at 10th weeks of gestation. Intracervical
fluid collection still exists
rate is low due to early ultrasonographic diagnosis. Thus
diagnosing the condition as early as possible is of great
importance.[1,8,9] Gestational age at the time of diagnosis
ranges from 5 to 8 weeks. [10] In ART pregnancies,
awareness of the possibility of a heterotopic pregnancy
and its sonographic appearance plays an important role in
accurate and timely diagnosis and successful management
to help avert potentially fatal consequences. Hence, a high
index of suspicion is mandatory for early diagnosis. The
presence of an intrauterine gestation does not exclude the
possibility of a concomitant extrauterine pregnancy. Careful
evaluation of the cervical canal as well as the uterine horns
and the adnexa should be a critical part of the ultrasound
examination even when an IUP has been confirmed. This
is true especially when the blood hCG level is much higher
than expected. The cervical component of the HCP can
be mistaken for other pathologies: Incomplete abortion,
normal pregnancy with low uterine implantation, EP in a
cesarean section scar, nabothian cyst, and cervical mass.
The gestational sac in the cervix is typically eccentrically
located and is usually round and similar to a normal
pregnancy. It may, however, become elliptical or flattened,
thus making diagnosis difficult.[11] Cardiac activity is
basically pathognomonic.
In general, uterine anomalies are a well‑known predisposing
factor for ectopic pregnancies. [12] For heterotopic
pregnancies, transferring four or more embryos during
ART is a defined risk factor.[13] However, no single risk
factor, laboratory test or combination of these is sensitive
or specific enough to predict the occurrence of an HCP.
In theory, it is possible that after pushing the plunger
during ET, the existence of sticky mucus may cause one
of the embryos to stuck and slide back to the canal. Thus,
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Isikoglu: Treatment of heterotopic cervical pregnancy
remnants of cervical mucus may be a risk factor. This
theory is supported by the fact that almost all of the HCPs
are the result of ART. Thus, we propose that removing
the cervical mucus effectively may be the only preventive
measure to try to avoid this extremely rare entity.
In general, the aims of the treatment are the protection
of a coexisting IUP, the minimization of blood loss, and
fertility preservation. Since HCP is a rare entity, there are
not established standard protocols but several options exist
for the management.
Expecting spontaneous expulsion of the cervical
component
Fruscalzo et al. reported a case of a simultaneous
nonviable cervical pregnancy and viable IUP diagnosed
in the 13th gestational week. Expulsion of the cervical
pregnancy occurred three weeks later followed by
profound hemorrhage, conservatively managed with
cervical curettage and stitches. Spontaneous abortion of
the intrauterine fetus ensued few hours later.[14]
Uterine artery ligation or angiographic arterial
embolization
It may result in the radiation of the viable IUP, and
influence on endometrial receptivity, which could decrease
future fertility.[15] Uterine artery ligation and embolization
may be options when the IUP is not of concern.
Local KCL administration
Embryo reduction procedure by ultrasound‑guided
transvaginal injection of potassium chloride into the ectopic
component is another treatment modality. The ongoing
pregnancy might be complicated by persistence and even
enlargement of remaining trophoblastic tissue, leading to
obstetric hemorrhage and emergency intervention.[16]
Local methotrexate administration
Risk of systemic adverse effects such as thrombocytopenia,
leukopenia, elevated serum liver enzymes, and especially the
teratogenic effect, should be taken into consideration.[17]
Systemic methotrexate administration
It is an option only when the aim is not to preserve the IUP.
Surgical treatment
Surgical treatments include suction evacuation,[18] cervical
curettage with or without cerclage and Foley catheter
insertion.[19,20] Jozwiak et  al. attempted a novel approach
using hysteroscopic removal followed by roller‑ball
coagulation of the bleeding sites. The pregnancy then
continued successfully to be terminated by a near‑term
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cesarean section without any abnormal bleeding. [21]
Obviously, this approach needs an intact internal cervical
os to keep the intrauterine part unaffected.
Foley catheter insertion and cervical cerclage seem
universal salvage maneuvers to stop early or late bleeding
whichever technique is used to terminate the cervically
located gestation. Mashiach et al. proposed elective cervical
Shirodkar cerclage itself as the treatment modality of
choice for cervical and HCP.[19]
Kim et al. reported a case of HCP successfully treated by
transvaginal embryo reduction with ovum forceps, the
outcome of the IUP was a healthy term delivery.[6]
Nonsurgical treatments may not be rationale in women
who are hemodynamically unstable. Laparotomy may rarely
be needed if any even in women with profound bleeding.
In the literature, the attempt to preserve the IUP was
successful in 83% (25 out of 30) of the cases resulting in
24 live births; 58.3% (7 out of 12) of the cases developed
serious complications when complete evacuation of the
cervical component was not performed, regardless of
the initial procedure, while 91.7% (11 out of 12) whose
treatment included complete evacuation of the cervical
pregnancy had no major complications.[6]
In our two cases, we performed simple embryo aspiration
without complete evacuation of the conceptus. In one
case, the pregnancy resulted in healthy delivery while
the second case was terminated due to chorioamnionitis
followed by preterm premature rupture of membranes.
In the existing literature, another HCP case managed
similarly via simple aspiration without any complication
and delivery of a healthy infant at 35 weeks was reported
by Cho et al.[22]
This minimally invasive approach in an hemodynamically
stable patient can be considered in the management of a
first‑trimester HCP. However prior to this procedure, the
patient should be counseled about the risk of the potential
postoperative complications including bleeding, abortion
of the IUP, cervical mass infection which may lead to
premature rupture of the membrane, and postpartum
bleeding and severe bleeding leading to potential need for
emergency procedures including even hysterectomy.
For cervical pregnancies without a simultaneous intrauterine
gestation, complete evacuation of the pregnancy or
systemic methotrexate administration seem better options
due to the risk/benefit ratio of the reduction procedure.
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Isikoglu: Treatment of heterotopic cervical pregnancy
Fortunately, there is a remarkable improvement in the
prognosis of cervical gestations including HCP. Tools for
early diagnosis especially provide enough time for elective
procedures. Nevertheless, one should be well‑prepared
prior to do any intervention to avoid inadvertent
complications in this potentially life‑threatening entity.
CONCLUSION
There is no universally accepted treatment modality mainly
due to the limited number of cases in the literature. Hence,
the management should be individualized based on the
hemodynamic status of the patient, technical availability
of the facility, and the skills of the surgeon.
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Source of Support: Nil, Conflict of Interest: None declared.
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