JBRA Assisted Reproduction 2019;23(4):434-438

doi: 10.5935/1518-0557.20190036 Case report

Cervical ectopic pregnancy after in vitro fertilization: case report

successfully treated with cervical electric aspiration
Jefferson Drezett1, Dinah Marques1, Roberta Ottoboni1, Artur Dzik1, Mario Cavagna1

1
Reproduction Department of Pérola Byington Hospital

ABSTRACT different interventions such as cervical suture, cerclage

Cervical ectopic gestation is a serious and potentially with uterine curettage, cervical balloon tamponade, uter-
lethal condition considered exceptional in in vitro fertiliza- ine artery embolization, methotrexate, potassium chloride
tion. Early diagnosis is critical to successful treatment and injection or hysteroscopic resection (Jozwiak et al., 2003;
preservation of fertility. We report a rare case of cervical Chen et al., 2001; Prorocic & Vasiljevic, 2007; Bennett et
pregnancy after in vitro fertilization and embryo transfer, al., 1993).
successfully treated exclusively with electrical aspiration. Despite these alternatives, the diagnosis is rarely early
Case report: A 36-year-old patient attended the Depart- enough so that the treatment can be done through aspi-
ment of Human Reproduction at Pérola Byington Hospital ration of the pregnancy without the occurrence of intense
in 2015 due to primary infertility with no apparent cause bleeding or the need for emergency hysterectomy. Thus,
for seven years. Subjected to ovulation induction with re- the objective of this article is to describe a rare clinical
combinant depot FSH and GnRh analogue, triggered with case of cervical ectopic pregnancy after in vitro fertilization
chorionic gonadotropin. It evolved with the collection of (IVF) with embryo transfer (ET), successfully treated with
ten oocytes and transfer of two embryos, with cryopreser- exclusive electric aspiration of the cervical canal. Because
vation of the remaining ones. The control with serial ultra- it was a case report, it was not necessary to submit to
sound showed a gestational sac in uterine cervix topogra- the Research Ethics Committee or to adopt a Free and In-
phy, indicating gestation of six weeks, confirmed after 24 formed Consent Form.
hours by a second operator. The treatment was success-
fully performed by electric aspiration with an EasyGrip® CASE REPORT
cannula of 6 mm in diameter, without the occurrence of A 36-year-old patient admitted to the Human Repro-
hemorrhage or the need for other procedures. Early diag- duction Department of Pearl Byington Hospital in March
nosis allowed successful conservative treatment with only 2015 for primary infertility seven years ago. Menarche at
cervical aspiration. The literature review confirms the rarity eleven years, with regular cycles of four days and inter-
of the case, but does not indicate consensus on the best val of 28 days. For seven years, she had had three inter-
treatment of cervical ectopic pregnancy. course sessions per week without the use of a contracep-
tive method. A history of hyperprolactinemia with nuclear
Keywords: Ectopic pregnancy, Cervix uteri, In vitro magnetic resonance without torsal seizures in 2012, treat-
fertilization, Multifetal pregnancy reduction ed with cabergoline for two years with normalization of
prolactin levels.
Physical examination at admission showed good gen-
INTRODUCTION eral and nutritional status. Transvaginal ultrasonography
indicated 43 cc uterus, 7 mm intramural myoma and nor-
Cervical pregnancy is a rare and potentially lethal form
mal volume and appearance ovaries. Oncotic colpocitolo-
of ectopic gestation, with incidence between 1:2,500 and
gy without neoplastic alterations or inflammatory condi-
1:12,000 clinical pregnancies (Anev et al., 2013; Jozwiak
tion. Hysterosalpingography showed patent tubes with no
et al., 2003; Lin et al., 2013). The occurrence of cervical
abnormalities. Hemogram with Hb 10.2 g/dl, leukocytes
pregnancy has increased in recent decades with the in-
5,160 thousand/mm3, 542 thousand platelets; creatinine
creasing use of assisted reproductive techniques, although
0.7 mg/dl; glucose 99 mg/dl; TSH 1.65 mIU/L; Free T4
the causes are not sufficiently clarified (Anev et al., 2013;
0.82 ng/dL; prolactin 15.6 ng/dL; estradiol 34 pg/ml; CA
Chen et al., 2001; Lesny et al., 1999; Weyerman et al.,
125 14.8 U/mL. Serologies for hepatitis B, hepatitis C,
1989).
syphilis, HIV and rubella were negative.
In the clinical diagnosis of cervical pregnancy, the uter-
The patient underwent ovulation induction, initiated
us may be hourglass-shaped, indicating disproportion be-
on the third day of the cycle with recombinant depot FSH
tween the size of the body and the cervix. In a few cas-
(Elonva® 150 mcg) and GnRh analog (Orgalutran® 0.25
es it is possible to visualize the gestational content in the
mg) for six days, with follow-up of follicular development
specular examination when there is dilatation of the cervix.
by serial transvaginal ultrasonography. On the second day
These findings, however, correspond to the late diagnosis,
of the cycle, the right ovary had four antral follicles, the left
generally after the 10th week of gestation, when there is
ovary with six antral follicles and an endometrial echo of
a greater risk of hemorrhage and hypovolemic shock, or
5.9 mm. At the end of the induction showed three follicles
the need for emergency hysterectomy (Chen et al., 2001).
above 18 mm in the right ovary and another five follicles
Ultrasonography is essential to confirm early cervical
of similar measurements in the left ovary. Triggered with
pregnancy, significantly increasing the possibility of pre-
chorionic gonadotrophin (Choriomon® 5,000 IU).
serving the reproductive capacity of the woman. The treat-
Subjected to follicular aspiration with total collection
ment of cervical ectopic pregnancy is admittedly difficult
of ten oocytes. A conventional IVF was performed, with a
and there is no consensus on the best approach (Lesny et
total of nine embryos, and two embryos were transferred
al., 1999). The success of the treatment is reported with
to D3 with a Sydney catheter, without the use of tweezers

Received December 10, 2018

434
Accepted May 02, 2019
 Cervical ectopic pregnancy - Drezett, J. 435

for neck extension, scarce blood in the catheter and ab- The etiology of cervical gestation after IVF-ET is poorly
sence of embryo retained after transfer. Seven embryos understood. It is believed to be related to the exacerba-
were cryopreserved. tion of contractions of the junctional zone of the uterus
Patient presented dosage of beta hCG 4,815.93 IU/mL in the luteal phase as a consequence of the elevation of
on the 26th day after ET. At 33 days ET had vaginal bleed- progesterone, producing a similar effect to that occurring
ing. Transvaginal ultrasonography showed cystic image on the tubal motility. It is possible that the uterine fundus
measuring 4.9 mm in the cervix, compatible with gesta- stimulation provoked by the probe during ET also changes
tional sac with 3 mm embryo, indicating cervical gestation the contractility of the junctional zone. In addition, wom-
of 6 weeks (Figures 1 and 2). It was observed peripheral en who attended other forms of ectopic pregnancy had a
and parietal neovascularization with high resistivity index. higher peak of estradiol after ET (Lesny et al., 1999).
The test was repeated after 24 hours by another operator, Authors such as Bennett et al. (1993) and Yu et al.
confirming the diagnosis. (2014) accept these hypotheses, although they warn that
Laparoscopic emptying of the cervix with a 6 mm di- women who undergo assisted reproduction procedures and
ameter Karman cannula (EasyGrip® cannula 6 mm) was who undergo cervical ectopic pregnancy often have other
performed without the use of ultrasonography. No cervi- recognized risk factors, such as previous uterine instru-
cal canal tamponade was required due to scarce cervical mentation, uterine and cervical anatomical abnormalities,
bleeding at the end of the operative period. Patient was Asheman's syndrome, uterine fibrosis, chronic endometri-
kept hospitalized for 24 hours for strict observation of vag- tis or endometrial atrophy.
inal bleeding. She was discharged in good clinical condi- ET timing does not appear to be associated with an
tion, without vaginal bleeding and without the need for increased risk of cervical ectopic pregnancy (Lesny et al.,
other interventions. She returned after a week to review 1999). However, authors suggest that the difficulty of ET
the procedure, without vaginal bleeding and with normal or manipulation of the uterine cervix increases the risk
ultrasound examination. of ectopic pregnancy, either through the use of dilators,
rigid probes for wire transfer or guidewire, or neck elon-
DISCUSSION gation clamps (Bennett et al. 1993; Lesny et al., 1999;
Chen et al., 2001). Considering these possibilities, Yovich
The review of the literature in the database of the US
et al. (1985) recommend that the ET catheter be insert-
National Library of Medicine National Institute of Health
ed 55 mm routinely. In this case report, the patient had
(PubMed) using the descriptors ("Fertilization in Vitro"
no known risk factors for cervical ectopic pregnancy, no
[Mesh]) and "Pregnancy, Ectopic" and "Cervix Uteri" Mesh]
dilation or elongation of the cervix was used, and the ET
results in 25 articles published in the last 32 years, indicat-
presented no difficulty.
ing the rarity of cervical ectopic pregnancy after IVF.
The literature reports 12 cases of exclusive cervical
The literature indicates a frequent association of cer-
ectopic pregnancy after IVF-ET (Weyerman et al., 1989;
vical ectopic pregnancy after IVF-ET with heterotopic
Bennett et al., 1993; Ginsburg et al., 1994; Saliken et al.,
pregnancy. This form of ectopic pregnancy is defined by
1994; Pattinson et al., 1994. Two cases of twin cervical ec-
concomitance with another intrauterine pregnancy, a rare
topic pregnancy, rarer, are reported by Anev et al. (2013)
event in spontaneous pregnancies estimated at 1: 30,000
and by Aboulfoutouh et al. (2011). In all other cases, cer-
pregnancies. However, in the last three decades this rate
vical ectopic pregnancy after IVF-ET was heterotopic. Oth-
has been increasing significantly with assisted reproduc-
er exceptional situations are described by Lin et al. (2013),
tion techniques (Jozwiak et al., 2003; Lin et al., 2013).
in case of triple heterotopic pregnancy after IVF-ET, with

Figure 1. Transvaginal ultrasonography identifying the ectopic pregnancy presenting gestational

sac in cervical topography and empty uterine cavity

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Case report 436

Figure 2. Detail on transvaginal ultrasonography of cervical ectopic gestation

topical, tubal and cervical embryo, and by Davies et al. al. (2001) also chose the aspiration of cervical pregnancy
(1990), who describe cervical heterotopic gestation in a after IVF-ET, but associated the suture of the neck to pre-
patient with previous cervical ectopic pregnancy. vent hemorrhage, preserving the topical pregnancy until
According to Chen et al. (2001), hysterectomy re- the term.
mained until the early 1980s as the main treatment of Some authors adopt more complex treatments in more
cervical ectopic pregnancy, definitively compromising the adverse circumstances. Tsakos et al. (2015) report cases
woman's reproductive future. Ultrasonography changed of heterotopic cervical pregnancy with live embryos after
this scenario, reducing maternal mortality in cases of cer- oocyte donation IVF-ET, successfully treated with cervical
vical ectopic pregnancy from 40% to less than 10%. When aspiration followed by placement of Foley catheter and cer-
performed before the 10th week of pregnancy, ultrasonog- clage. Topical pregnancy followed without complications
raphy makes the early diagnosis of cervical ectopic preg- with elective cesarean delivery at week 38. In another
nancy. This allows more effective interventions that sig- case, Prorocic & Vasiljevic (2007) were able to preserve an
nificantly reduce the risk of bleeding and death, as well as intrauterine twin pregnancy by treating cervical pregnancy
maintaining the reproductive capacity of women (Bennett with aspiration and injection of hypertonic solution of KCL
et al., 1993). Magnetic resonance imaging has rarely been after ligation of descending cervical branches of the uterine
used in the propaedeutics of cervical ectopic pregnancy arteries.
and should be considered, as advocated by Pattinson et al. The exclusive use of the KCL injection for embryo re-
(1994) and Ginsburg et al. (1994). duction in a case of cervical ectopic pregnancy was an
In the last decades conservative treatments have been alternative of Carreno et al. (2000), resulting in topical
described with success, mainly in the heterotopic gesta- gestation up to the 36th week with a healthy newborn.
tions in which it is intended to maintain intrauterine ges- Honey et al. (1999) used the same strategy for cervical
tation. Only one case of spontaneous abortion of cervical embryo reduction, preceded by selective fluoroscopic em-
heterotopic pregnancy with normal follow-up of intrauter- bolization of the uterine arteries for prophylaxis of cervical
ine pregnancy is reported by Livingstone et al. (2000). hemorrhage. Although they have been successful in the
Anev et al. (2013) believe that the method should be de- treatment of cervical pregnancy, adverse events have not
cided together with the woman, considering her desire for allowed the evolution of topical pregnancy until the term.
future pregnancy and the experience of the medical staff. Hysteroscopic resection was alternative to Jozwiak
Retrospective study by Yu et al. (2014) with 25 heterotopic et al. (2003) to treat cervical heterotopic pregnancy and
pregnancies of different types showed that most cases of maintain intrauterine pregnancy to term successfully. The
heterotopic cervical pregnancy were treated with local in- use of methotrexate for the treatment of cervical hetero-
jection of methotrexate, combined or not with the injection topic gestation is described by authors such as Bratta et
of potassium chloride. al. (1996), Hsieh et al. (2004), Aboulfoutouh et al. (2011),
Cho et al. (2007) reported a case of aspiration of cervi- Sánchez-Ferrer et al. (2011) and Tsakos et al. (2015). The
cal ectopic pregnancy after 7 weeks IVF-ET guided by ultra- association of methotrexate with embolization of the uter-
sonography, maintaining the gestation until the 35th week. ine arteries and cervical aspiration was adopted by Sán-
Similar treatment was adopted by Hsieh et al. (2008), with chez-Ferrer et al. (2011).
transvaginal aspiration of the cervical embryo guided by The few cases of exclusive cervical ectopic pregnancy
ultrasonography preserving intrauterine gestation. Chen et after IVF-ET described in the literature were treated with

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 Cervical ectopic pregnancy - Drezett, J. 437

different therapeutic approaches. Uterine artery emboliza- Bennett S, Waterstone J, Parsons J, Creighton S. Two cases of
tion was employed by Saliken et al. (1994) as a preopera- cervical pregnancy following in vitro fertilization and embryo
tive measure. Subsequently, Yu et al. (2009) opted for the transfer to the lower uterine cavity. J Assist Reprod Genet.
same procedure before cervical emptying, also achieving 1993;10:100-3. PMID: 8499672. DOI: 10.1007/BF01204450
success. Two cases of cervical gestation after IVF-ET were
reported by Bennett et al. (1993), both successful with as- Bratta FG, Ceci O, Loizzi P. Combined intra-uterine and
piration of pregnancy followed by tamponade of the cervix cervical pregnancy treated successfully with methotrexate.
with balloon. Int J Gynaecol Obstet. 1996;53:173-4. PMID: 8735300
Other forms of treatment of cervical ectopic pregnan- DOI: 10.1016/0020-7292(96)83568-5
cy are described. Hsieh et al. (2004) opted for the use of
methotrexate and intracervical injection of vasopressin. In Carreno CA, King M, Johnson MP, Yaron Y, Diamond MP,
an unusual case, Anev et al. (2013) describe a diamniotic Bush D, Evans MI. Treatment of heterotopic cervical and
and monochorionic twin cervical ectopic pregnancy after intrauterine pregnancy. Fetal Diagn Ther. 2000;15:1-3.
single ET, diagnosed at week 6 and treated with the com- PMID: 10705207. DOI: 10.1159/000020967.
bination of mifepristone and systemic methotrexate, fol-
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(2011) also reported a case of twin cervical ectopic preg-
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et al. (1997) have been successful in employing exclusive
S0015-0282(01)01746-0.
methotrexate to treat cervical ectopic pregnancy. Similar
procedure was adopted by Piccioni et al. (2015), which
made possible later pregnancy to term. Cho JH, Kwon H, Lee KW, Han WB. Cervical heterotopic
Few cases of second trimester cervical pregnancy pregnancy after assisted reproductive technology success-
are described. The first case of second trimester cervical fully treated with only simple embryo aspiration: a case
pregnancy after IVF-ET was described by Weyerman et report. J Reprod Med. 2007;52:250-2. PMID: 17465300.
al. (1989), completed at the 26th week of gestation af-
ter hysterectomy, with a newborn of 830 grams. Pattinson Davies DW, Masson GM, McNeal AD, Gadd SC. Simulta-
et al. (1994) report the treatment of this condition with neous intrauterine and cervical pregnancies after in vitro
embolization of the uterine arteries followed by emptying fertilization and embryo transfer in a patient with a his-
of the cervix and placement of an intracervical balloon. tory of a previous cervical pregnancy. Case report. Br J
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CONCLUSION tion--a successful treatment to control bleeding cervical
Cervical ectopic pregnancy after IVF-ET is a rare condi- pregnancy with a simultaneous intrauterine gestation.
tion. The different approaches found in the literature indi- Hum Reprod. 1999;14:553-5. PMID: 10100008. DOI:
cate that there is no consensus on the best treatment. This 10.1093/humrep/14.2.553
report is the only case of successful treatment of cervical
pregnancy after IVF-EF only with the use of cervical aspi- Hsieh BC, Lin YH, Huang LW, Chang JZ, Seow KM, Pan
ration. The experience of different authors is unanimous in HS, Hwang JL. Cervical pregnancy after in vitro fertilization
considering the fundamental routine ultrasonography after and embryo transfer successfully treated with methotrex-
IVF-ET for the early diagnosis of exclusive or heterotopic ate and intracervical injection of vasopressin. Acta Obstet
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CONFLICT OF INTEREST Hsieh BC, Seow KM, Hwang JL, Lin YH, Huang LW, Chen HJ,
The authors have no conflict of interest to declare. Tzeng CR. Conservative treatment of cervico-isthmic het-
erotopic pregnancy by fine needle aspiration for selective
Corresponding Author: embryo reduction. Reprod Biomed Online. 2008;17:803-5.
Jefferson Drezett PMID: 19079964. DOI: 10.1016/S1472-6483(10)60408-7
Reproduction Department
Pérola Byington Hospital Jozwiak EA, Ulug U, Akman MA, Bahceci M. Success-
São Paulo - SP - Brazil ful resection of a heterotopic cervical pregnancy result-
E-mail: [email protected] ing from intracytoplasmic sperm injection. Fertil Steril.
2003;79:428-30. PMID: 12568859. DOI: 10.1016/S0015-
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