Ectopic pregnancy in uncommon implantation sites

Sanaa Badr
a
, Abdel-Naser Ghareep
b,
*
, Lamya M. Abdulla
b
, Rabei Hassanein
c
a
Radiology Department of Women Hospital, Doha, Qatar
b
Radiology Department, Al-Azhar University, Cairo, Egypt
c
Obstetric-Gynecology Department, Al-Azhar University, Cairo, Egypt
Received 7 June 2012; accepted 24 October 2012
Available online 27 November 2012
KEYWORDS
Ectopic pregnancy;
Uncommon implantation
sites
Abstract The majority of ectopic pregnancies are located within the fallopian tube. Nevertheless,
pregnancies have been reported to implant in the cervix, ovary, interstitial tubal segment, and at
various intra-abdominal sites. The diagnosis and treatment of these unusual implantation sites pre-
sents a challenge for clinical as well as radiological diagnosis and there is a tendency to overlook its
possibility. In this study, we attempt to summarize the current data regarding diagnosis and optimal
treatment of these unusual ectopic pregnancies from our experience with six unusual types of ecto-
pic pregnancies from the Women Hospital, Doha, Qatar.
2012 Egyptian Society of Radiology and Nuclear Medicine. Production and hosting by Elsevier B.V.
All rights reserved.
1. Introduction
Ectopic pregnancy is among the leading causes of mortality
among pregnant women (1). Although the incidence of ectopic
pregnancy is estimated to be between 1% and 2%, the majority
(95%) of these pregnancies are located in isthmic/ampullary/
mbrial (extracornual) portion of the fallopian tube (2,3).
However, about 5% pregnancies also occur implanted in the
cervix, ovary, previous cesarean scar, interstitial portion of
the fallopian tube and abdomen as well as angular ectopic
pregnancy and heterotopic pregnancy (intrauterine pregnancy
(IUP) + ectopic) (4) (Fig. 1). The relative infrequency of these
implantation sites makes the study of treatment efcacy
difcult (5). Advances in ultrasound technology and operator
expertise have provided the capability to visualize ectopic preg-
nancy at an unusual location at its earliest stage (6). Although
routine two-dimensional ultrasound can be suggestive, 3D
ultrasound is highly accurate in diagnosis. From the Women
Hospital, Doha, Qatar, we illustrate and discuss six unusual
types of ectopic pregnancies, heterotopic pregnancy, scar preg-
nancy, interstitial pregnancy, cervical pregnancy, abdominal
pregnancy and ovarian pregnancy.
1.1. Case No. 1
The patient was 39 years old, gravida 3 para 1 + 1. The patient
was admitted through the emergency department with a missed
period and moderate abdominal pain. Patient was examined,
her vitals were stable, and ultrasound was requested. There
is an intrauterine pregnancy with extra uterine left sided tubal
pregnancy, so decision was taken for admission. In the ward,
the patient was stable with sudden left sided pain. Abdominal
examination shows abdomen is soft and lax with mild tender-
ness. On the 2nd day patient is well, ambulating, no abdominal
*
Corresponding author.
E-mail address: nasergharib@yahoo.com (A.-N. Ghareep).
Peer review under responsibility of Egyptian Society of Radiology and
Nuclear Medicine.
Production and hosting by Elsevier
The Egyptian Journal of Radiology and Nuclear Medicine (2013) 44, 121130
Egyptian Society of Radiology and Nuclear Medicine
The Egyptian Journal of Radiology and Nuclear Medicine
www.elsevier.com/locate/ejrnm
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0378-603X 2012 Egyptian Society of Radiology and Nuclear Medicine. Production and hosting by Elsevier B.V. All rights reserved.
http://dx.doi.org/10.1016/j.ejrnm.2012.10.006
pain. No vaginal bleeding. Ultrasound is conrmed, she has a
viable heterotopic pregnancy of 7 weeks (Fig. 2) and she
posted to laparoscopy.
Laparoscopy conrmed a left sided viable tubal pregnancy
involving the whole left tube and impending rupture. The left
tube (almost all) was cauterized and cut. The pregnancy was
taken out through an endo-bag and sent for histopathology.
The patient was discharged on the 3rd day with stable vital
signs and ultrasound was done to conrmed intrauterine pul-
sating pregnancy.
1.2. Case No. 2
A 25 year old patient, G2P1. She has previous CS. She is
5 weeks amenorrhea. Patient was admitted from the emer-
gency department as referred from a private hospital, where
she was diagnosed as ectopic pregnancy in the CS scar and
was given methotrexate injection. By examination, she was sta-
ble; abdomen was soft, lax, no tenderness and pelvic examina-
tion not done. BHCG (beta human chorionic gonadotropin)
done in private hospital was usually normal. Patient was
admitted in the ward, at night BHCG 7929 IU, high liver
function tests and normal CBC, Hb and PLT count.
Plan: observation, vital sign chart and repeat ultrasound in
the next day.
Ultrasound shows: gestational sac seen eccentrically located
in the lower uterine segment in the site of the previous LSCS
(Fig. 3). GSD = 1.1 cm showing a yolk sac and tiny fetal node
(CRL: 2.0 mm). No fetal cardiac activity seen or recorded.
On the next day, patient still vitally stable with a drop of
high liver function. We followed her on 13/09/2009 in the
emergency department: BHCG = 7870. Patient was dis-
charged with an appointment after 1 week and she was advised
if any bleeding or severe pain must come to emergency depart-
ment. After 1 week, the patient was seen in the clinic with
BHCG 16/9/2009 and 23/09/2009 and she was instructed for
BHCG weekly till it will become negative. Last BHCG on
26/10/2009 = 0.61 IU which is negative and the patient was
discharged from the clinic.
1.3. Case No. 3
A 32 year old female G3P1 + 1 in 8 weeks of amenorrhea
with previous CS admitted through the emergency department
with a mild brownish discharge and patient admitted to ward
planned for observation. O/E, she is vitally stable, abdomen is
soft, lax, no mass or tenderness.
Ultrasound (2D & 3D) was done and shows a non-viable fe-
tal pole within the gestational sac that is eccentrically located in
the right fundal side with the rimof myometrial tissue surround-
ing the pregnancy sac right interstitial pregnancy (Fig. 4).
Methotrexate was given with serial BHCG for follow up.
Serial BHCG: 23/05/2011 9325 IU 25/05/2011 4756 IU
So decision was taken to discharge her and she was in-
structed about all complications which can happen. The pa-
tient came for follow up in the outpatient clinic on 15/06/
2011. She was well, no pain or bleeding and BHCG
914 IU. So the patient was instructed to do BHCG weekly till
it will become negative. BHCG in 18/07/2011 0.16 IU
negative.
1.4. Case No. 4
A 26 year old lady, Primigravida, IVF pregnancy. ET = 4/2/
2008, so she is 9 weeks on 5/3/2008. She was admitted through
the emergency department, complaining of backache with no
bleeding. Routine investigation requested including blood tests
and ultrasound was done.
Ultrasound shows a well formed GS with surrounding
echogenic rim centrally located in the cervical canal with yolk
sac and embryo without cardiac pulsation and the endometrial
cavity is empty and diagnosed as cervical pregnancy. The
GSD = 1.99 cm and CRL = 0.28 cm corresponding to
6 weeks GA (Fig. 5). Patient was admitted to the ward, deci-
sion was taken for methotrexate therapy.
O/E: she is vitally stable, mild abdominal; abdomen was
soft, lax and not tender. Pelvic examination was not done with
slight bleeding since 1 h.
Patient receives methotrexate on 6/3/2008 with dropping of
BHCG level.
Plan will be for observation, CBC and BHCG.
The patient was re-admitted on 27/03/2008 complaining
from lower abdominal pain. O/E: she is vitally stable, abdo-
men is soft, lax and not tender and PV was not done. The pa-
tient was kept under observation until 1/4/2008 then was
discharged with BHCG weekly until it will be negative with
ultrasound and Doppler follow up. BHCG came down and be-
came negative on 14/6/2008.
Serial follow up US shows a gradual decrease in the size of
the sac and CRL until the last follow up study after three
months showing the GS is markedly collapsed and only a small
remnant about 11 5 mm is detected (Fig. 6).
1.5. Case No. 5
A 36 year patient, G1P0, IVF twin pregnancy (Dichorionic
Diamniotic)
Embryo transfer (ET): 02/07/08.
EDD: 24/03/09.
Known to be gestational diabetic on diet.
Current pregnancy:
The patient had her 2nd successful trial of IVF pregnancy
which is twins pregnancy (DiDi).
Fig. 1 Diagrams showing the usual and unusual sites of ectopic
pregnancy.
122 S. Badr et al.
She started to follow in Women Hospital out patient
clinic from 22 weeks where she had 3 visits to clinic at
223435 weeks.
Apart from gestational diabetes on diet, her other inves-
tigations were normal including US.
First US scan done at 23 weeks: compatible with date with
no anomalies. Then that scan was followed by four other
scans, all were found to be with normal growth and normal
Doppler.
Last scan was on 15/02/09, (DiDi), normal growth, nor-
mal liquor and Doppler for both fetuses, 1st cephalic (2.1)
kg and 2nd transverse (2.2) kg.
On 05/03/09 the patient came to emergency complaining of
watery vaginal discharge with lower abdominal pain and leak-
ing conrmed in emergency by speculum Ex. PV: 3 cm, 80%
effaced, cephalic, -3 stations, so patient admitted to labor
room (LR).
In LR: bed side US showed both fetuses cephalic and
over 3.5 h the patient progressed nicely to fully dilated cer-
vix under epidural analgesia and remained fully dilated for
2 h. The 1st twin delivered was female, cephalic with AP-
GAR 910 weighing 2295 gm then the cervix reformed
where the 2nd twin lie transversely. So, decision taken for
emergency CS.
On laparotomy;
There was gestational sac outside the intact uterus.
The sac opened through a thin area where no placenta is.
Baby boy delivered with APGAR score 910, 2265 gm.
Fig. 2 US showing heterotopic pregnancy, intra and extra uterine viable fetus 7 weeks, with laparoscopic picture of the patient.
Ectopic pregnancy in uncommon implantation sites 123
After delivery of baby, the placenta was found attached to
cecum, sigmoid colon, back of uterus till fundus & Rt.
broad ligament (Fig. 7).
The sac has a vascular pedicle which clamped, divided and
removed completely with placenta.
Both maternal and fetal conditions were clinically and
hemodynamically stable in the ward. The patient had
remarkable recovery.
1.6. Case No. 6
A 28-year-old lady, nullipara presented to the emergency
department with vaginal spotting and severe abdominal pain.
Qualitative pregnancy test was positive, so quantitative test
(BHCG) taken and it was 15531 IU.
Ultrasound revealed, normal size RVF empty uterus with a
large heterogeneous left adnexial mass around 10.6 6.8 cm
seen with turbid uid in the Douglas pouch (Fig. 8).
The patient is generally stable with abdominal tenderness
with re-bound tenderness, maximum on the left iliac fossa with
no rigidity.
Pelvic exam: RVF uterus, normal size, positive cervical
excision, so decision was taken for urgent laparoscopy.
During the procedure: There is moderate internal hemor-
rhage. Blood was evacuated using suction with inspection of
pelvis. There is a brown soft tissue material measuring
10 7 cm with a small area of oozing. Mass excised was taken
out using the endo-bag and sent for histopathology. Suction
irrigation of the pelvis and homeostasis were done. The patient
was discharged in the next day with good general condition.
BHCG = 6008 IU. She had an appointment in the clinic after
2 weeks to check histopathology and BHCG (become729 IU
after 2 weeks).
Histopathology report:
1. Left ovary excision consistent with ectopic pregnancy
(chronic villi seen).
2. Left fallopian tube cystectomy consistent with para-tubal
cyst (hydatid of morgagni).
2. Discussion
Ectopic pregnancy at any location is a serious problem due to
signicant maternal morbidity and mortality. Although the
unusual implantation sites are much less frequent, these must
also be considered (7).
2.1. Heterotopic pregnancy
A rare type of multiple pregnancies involving one viable preg-
nancy in the uterus and the other implanted elsewhere as an ec-
topic pregnancy (our illustrated case No. 1 both are viable).
Fig. 3 Showing non-viable ectopic pregnancy in lower cesarean section scar (LSCS).
124 S. Badr et al.
Heterotopic pregnancies occur with rates less than one in
30,000 naturally occurring pregnancies (as in our case No.
1), and one in 100 couples who conceive through assisted
reproduction (8).
The risk of both ectopic and heterotopic gestations in-
creased in: (a) assisted reproduction techniques due to super
ovulation, (b) patients with previous tubal surgeries, (c) persis-
tent or rising BHCG levels after an induced/spontaneous abor-
tion, (d) when the uterine fundus is larger than for menstrual
dates, (e) when more than one corpus luteum is present in a
natural conception (9,10). Most commonly, the location of ec-
topic gestation in a heterotopic pregnancy is the fallopian tube
(as our case No. 1). However, cervical and ovarian heterotopic
pregnancies have also been reported (11,12). Intrauterine
Fig. 4 Showing eccentrically located GS in the right fundal side separated from endometrial cavity by myometrial tissue consistent with
right interstitial pregnancy.
Fig. 5 Abdominal and TV US with Doppler showing non-viable 6 weeks ectopic cervical pregnancy.
Ectopic pregnancy in uncommon implantation sites 125
gestation with hemorrhagic corpus luteum can simulate het-
erotopic/ectopic gestation both clinically and on ultrasound
(13). A high resolution transvaginal ultrasound with color
Doppler will be helpful as the trophoblastic tissue in the ad-
nexa in a case of heterotopic pregnancy shows increased ow
with signicantly reduced RI (9). As in our illustrated case
Fig. 6 The last follow up US and Doppler study (3 month) after methotrexate therapy showing only small remnant.
Fig. 7 Laparotomy of the patient showing (a) Intact posterior wall with membranous adhesion to rectum, (b) membranous attachment
to the sigmoid colon, (c) main feeding from broad ligament with intact anterior uterine wall.
126 S. Badr et al.
and according to patient presentation and condition, the treat-
ment of a heterotopic pregnancy is laparoscopy/laparotomy
for the tubal pregnancy (10).
2.2. Cesarean scar pregnancy
CS scar pregnancy is a rare form of ectopic pregnancy with an
incidence of 1:1800 pregnancies (14). It is associated with com-
plications such as uterine rupture and massive hemorrhage
requiring emergency hysterectomy (15). A report describes a
successful pregnancy till term after previous cesarean ectopic
pregnancy (16). Diagnosis of cesarean ectopic pregnancy can
be made easily if the sonologist is familiarized with the case
(17). In our case No. 2, the cesarean scar ectopic pregnancy
was diagnosed early by ultrasound. Presentation of the patient
often dictates the mode of treatment of this case. Systemic
methotrexate followed by dilatation and evacuation have been
described (18). Treatment by suction curettage has been de-
scribed in selected cases (19). Other treatment options include
uterine artery embolization (20), laparoscopic ligation of bilat-
eral uterine arteries followed by excision of ectopic mass (21).
As used in our illustrated case, asymptomatic and hemody-
namically stable patients are candidates for medical manage-
ment by methotrexate.
2.3. Interstitial ectopic pregnancy
Interstitial pregnancy occur in interstitial segment of the fallo-
pian tube that lies within the muscular wall of the uterus (22)
and accounts for up to 13% of all ectopic pregnancies (23).
Correct diagnosis of ectopic pregnancy can be quite difcult.
It requires accurate ultrasound interpretation. The diagnosis
relies heavily on ultrasound and potentially on laparoscopic
evaluation (24). Ultrasound frequently shows a thin rim of
myometrial tissue surrounding the ectopic pregnancy sac (25).
The interstitial line has been described as an echogenic line
extending into the corneal region and abutting the gestational
sac, and is highly specic for interstitial pregnancy (3). In our
case No. 3, the diagnosis was suggested on a 3D transvaginal
ultrasonography prior to development of any complication,
resulting in early management. The traditional treatment of
interstitial pregnancy has been cornual resection or hysterec-
tomy in cases with severely damaged uterus (26). However,
there are successful case reports of laparoscopic resection of
cornual pregnancies (27). Although the surgical management
of interstitial pregnancy has remained most common, metho-
trexate has been used as the rst-line treatment, we use this line
in our case No. 3. In an analysis of 13 case series of a total of 47
total cases treated with methotrexate, Fisch et al. concluded
that methotrexate is a safe option. However, there are reports
of high failure rates, and there is a need for proper patient selec-
tion and extended monitoring (28).
2.4. Cervical ectopic pregnancy
Cervical ectopic pregnancies account for less than 1% of all
pregnancies, with an estimated incidence of one in 2500 to
one in 18,000 (29,30). In the past, cervical ectopic pregnancy
was associated with signicant hemorrhage and was treated pre-
sumptively with hysterectomy. Improved ultrasound resolution
Fig. 8 Left adnexial mass with pelvic hemorrhage seen by US and laparoscopy.
Ectopic pregnancy in uncommon implantation sites 127
and earlier detection of these pregnancies have led to the devel-
opment of more conservative treatments that attempt to limit
morbidity and preserve fertility.
Cervical pregnancy is diagnosed by US according to the fol-
lowing criteria described by Hofmann and Timor-Tritsch (31).
1. Presence of gestational sac or placental texture dominantly
within the cervix.
2. No evidence of intrauterine pregnancy.
3. Visualization of an endometrial stripe (except in case of het-
erotopic pregnancy).
4. Hourglass uterine shape with ballooned cervical canal.
5. Sac with active cardiac motion below the internal Os for
viable pregnancy.
Color Doppler scanning is useful in differentiating between
inevitable miscarriage of a gestational sac in the cervix and true
cervical pregnancy. In true cervical pregnancy, Doppler studies
show characteristic patterns of trophoblast with high ow
velocity and low impedance, while in miscarriage the sac will
be mobile, with no Doppler evidence of blood ow(32,33). Cer-
vical pregnancies before 12 weeks, without fetal cardiac activity
and with lower serum BHCG levels seem more amenable to
conservative treatment (34,35). Most reports of successful con-
servative therapy involve the use of systemic chemotherapy in
combination with cervical evacuation and the use of hemostatic
techniques (36,37). Spitzer et al. (34) reported using curettage
followed by the injection of prostaglandin F2a to increase uter-
ine contractions, promote vasoconstriction, and therefore, re-
duce hemorrhage. In our case No. 4 we use the conservative
measures with systemic methotrexate and follow up as it be-
come the standard rst-line approach to treatment of women
who desire fertility preservation and the treatment was success-
ful and reached the full resolution.
2.5. Abdominal ectopic pregnancy
Abdominal pregnancy is an extremely rare event (1.4% of ecto-
pic pregnancies) that may be difcult to diagnose and aware-
ness of this condition is very important in reducing the
associated morbidity and mortality. Abdominal pregnancies re-
fer to those with extra uterine implantations in omentum, vital
organs, or large vessels. These pregnancies can go undetected
until an advanced gestational age and often result in severe
hemorrhage, disseminated intravascular coagulation, bowel
obstruction, and stulae (3840). Frequently, these pregnancies
are encountered with a viable fetus, which complicates their
management.
The diagnosis is often made using ultrasound and X-ray.
Sonographic features suggestive of abdominal pregnancy in-
clude empty uterus, absence of myometrium around the fetus,
oligohydramnios, abnormal fetal lie and poorly dened pla-
centa (41). Elevated serum alpha-fetoprotein has also been
associated with abdominal pregnancy. Diagnostic laparoscopy
may also be of value when there is a doubt about pregnancy
location (42). In some cases, the diagnosis is not made until
laparotomy (43), this occurs in our case No. 5 that diagnosed
by mistake as intrauterine twins. MRI holds promise as a diag-
nostic tool (43,44).
The optimal treatment of abdominal pregnancy is un-
known. It has been reported that management of the placenta
correlates well with maternal morbidity. When possible,
ligation of placental blood supply and removal should be at-
tempted to reduce maternal complications (45). Alternatively,
the umbilical cord may be ligated and expectant management,
arterial embolization, or methotrexate used to facilitate involu-
tion (42). However, leaving the placenta in situ may lead to
further complications such as infection, secondary hemor-
rhage, or intestinal obstruction (46). Olsen et al. (47) reported
laparoscopic management of a broad ligament pregnancy
without complication. Primary methotrexate has been at-
tempted for early gestations with minimal success (48).
2.6. Ovarian pregnancy
Ovarian pregnancy is one of the rarest varieties of ectopic
pregnancies accounting for 0.153% of all ectopic gestations.
Patients frequently present with abdominal pain and menstrual
irregularities. Preoperative diagnosis is challenging but trans-
vaginal sonography has often been helpful. A diagnostic delay
may lead to rupture, secondary implantation or operative dif-
culties. Therefore, awareness of this rare condition is impor-
tant in reducing the associated risks (49). The diagnosis
routinely is made by ultrasound appearance of a wide echo-
genic ring on the ovary, frequently with a yolk sac or fetal
parts. Ovarian pregnancies are often confused with corpus lut-
eum cysts (50). 3D ultrasound imaging has been used to distin-
guish ovarian pregnancy from corpus luteum cysts. It may be
useful to perform intraoperative ultrasound to distinguish an
ovarian pregnancy from an ovarian cyst (51). Our illustrated
case No. 6 diagnosed by US as tubal pregnancy and suspected
on laparoscopy and nally diagnosed by histopathology.
Ovarian pregnancy can be treated conservatively with single-
dose methotrexate. However, the preferred mode of treatment
is oophorectomy by laparotomy or laparoscopy, currently,
laparoscopic surgery is the treatment of choice.
3. Conclusion
Six unusual types of ectopic pregnancy were illustrated and
discussed in this article. These are heterotopic pregnancy (com-
bined intra- and extra uterine pregnancies), scar pregnancy,
interstitial pregnancy, cervical pregnancy, abdominal preg-
nancy and ovarian pregnancy. Ectopic pregnancies of unusual
location are encountered much less frequently, but are perhaps
more morbid. The use of advanced ultrasonography in combi-
nation with ultrasensitive serum b-hCG assays should lead to
early diagnosis. Early diagnosis and use of multiple modalities
can reduce morbidity and mortality in cases of ectopic preg-
nancy at unusual location.
The treatment of these unusual ectopic gestations may not
be as commonplace as treatment of tubal pregnancies, but with
early diagnosis and effective planning, their treatment can be
equally as effective.
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