Cesarean Scar Ectopic Pregnancy: A report of 2 cases

1
ABSTRACT:

A Cesarean scar pregnancy (CSP) is a rare and serious complication

of pregnancy, which consist of a gestational sac that is implanted

within the previous cesarean section scar. Due to the global increase

in cesarean sections as well as the improved diagnostic abilities,

incidence rates have continued to rise since the first reported case in

1978.

This condition often poses a diagnostic challenge, various articles

suggest different criteria to diagnose CSP using transvaginal

ultrasound. The case presented is a 31 year-old, G3P2 (2001) in her

7th weeks age of gestation with a previous history of cesarean delivery

presented with vaginal bleeding referred to our institution due to an

ultrasound finding of cesarean scar pregnancy. A repeat ultrasound

was performed confirming the diagnosis and managed successfully by

wedge resection of the cesarean scar pregnancy followed by

curettage.

These case reports aims to emphasize the importance of ultrasound

in the early prenatal diagnosis of this rare type of ectopic pregnancy,

which guides the multi-disciplinary team of health care providers as to

the management, careful planning and counseling of the patient and

its family.

2
INTRODUCTION:

Cesarean scar pregnancy is a rare form of ectopic pregnancy. The

etiology and pathophysiology are not yet fully understood, but the

main mechanism is theinvas ion of a microscopic tract within the CS

scar by the blastocyst as it implants (15). The reported incidence of

CSP ranges from 1:1800 to 1:2500 of all pregnancies, comprising

0.15% of all pregnancy with history of previous cesarean delivery

(6,7). Due to the increasing rate of cesarean delivery worldwide, this

led to the rising number of CS scar pregnancy.

The clinical presentation varies with at least 40% of detected cases

being asymptomatic, uterine rupture and potentially catastrophic

bleeding however can occur with the delayed detection (6). This paper

intends to emphasized the importance of early detection of cesarean

scar pregnancy by ultrasonography which is a mainstay in diagnosis

of CSP with reported sensitivity of 85% (6).

Since there is no standard protocol as to the management of CSP,

treatment should be individualized depending on the patients age,

desire for future fertility, number of children and the hemodynamic

status of the patient (2,5,7). This condition can be managed

expectantly, medical management using methrotrexate, surgical

management or in combination.

3
CASE REPORTS:

Case 1:

A case of a 31 year-old G3P2 (2001), on her 6 weeks and 4 days age

of gestation who came in due to vaginal spotting with an ultrasound

findings of Cesarean scar ectopic pregnancy. She had a history of

vaginal spotting for 1 week and sought consult at a private clinic

where pregnancy test was done and turned out positive. Transvaginal

ultrasound revealed a cesarean scar pregnancy hence patient was

advised transfer to a tertiary hospital.

She have 2 previous pregnancies all delivered full term via cesarean

section, but the first child died due to hydrocephalus. The patient had

minimal vaginal bleeding with no other accompanying signs and

symptoms such as abdominal pain. On physical assessment, patient

was ambulatory with stable vital signs, abdomen has a previous

midline scar, flat, soft, non-tender. On internal examination the cervix

is soft, closed, no cervical motion tenderness, uterus is small, no

adnexal mass nor tenderness with blood on examining finger.

A repeat transvaginal ultrasound scan showed an anteverted uterus

with a complex mass located within the myometrium at the anterior

lower uterine segment above the level of the internal cervical os within

the cs scar measuring 3.22 x 1.88 x 2.89 cm with a gestational sac

measuring 2.66 cm by mean sac diameter containing a yolk sac

measured 0.26 cm and an embryo with crown-rump length of 0.69 cm

equivalent to 6 weeks and 4 days age of gestation with fetal heart rate

4
of 133 bpm (Figure 1). The. The residual anterior myometrial

thickness measures 0.30 cm (Figure 4), the corpus luteum was seen

in the right ovary, the left ovary was normal. 3D scan of the uterus on

sagittal view shows a GS with a yolk sac and embryo at the anterior

myometrial wall with an empty endometrial cavity (Figure 2 & 3). No

adnexal mass was noted. All laboratory work-up are within normal

limits expect for the serum B-HCG which is elevated at 74,855

mIU/mL.

The patient was admitted with a diagnosis of G3P2 (2001) Cesarean

Scar pregnancy 6 weeks and 4 days by early ultrasound, unruptured,

Previous CS 2x for unknown type and indication (2012, 2017).

Family conferences was done to discuss management options to the

patient and her family, the patient decided for a conservative surgical

procedure to preserve her fertility, hence an ultrasound guided wedge

resection of the CS scar ectopic pregnancy followed by curettage,

adhesiolysis was done.

Prior to the surgical procedure a baseline ultrasound was done to

determine the exact location of the gestational sac (Figure 5).

Intraoperatively, the uterus is densely adherent to the bladder (Figure

6) hence another ultrasound was done to guide the surgeon in

adhesiolysis to prevent perforating the bladder and the thinned

anterior lower uterine segment. There is a 3x3 cm bulging mass

located at the previous cesarean scar and with the guidance of

ultrasound the puncture site was identified (Figure 7). A post

evacuation scan was done to confirm the complete removal of the

5
placental tissues (Figure 8&10), the surgery was unremarkable with

an estimated blood loss of 300 cc.

Post operatively, repeat quantitative serum beta HCG was done on

post-operative day 3 with a serum Beta HCG result of 13,380 mIU/mL,

the rest of the hospital stay were unremarkable hence patient was

discharged on the 6th post-op day. Histopathologic result revealed a

necrotic decidua and immature placental tissue (Figure 11).

The patient followed up after 1 week with no subjective complaints,

repeat ultrasound was done revealing a normal sized anteverted

uterus, thin endometrium with intracavitary fluid suggestive of blood

and both ovaries are normal (Figure 12). Patient was advised repeat

serum beta HCG and a repeat ultrasound 6 weeks postsurgery, but

did not follow-up as instructed.

Case 2:

A case of a 30 year-old G2P1 (1001), 7w1d by early ultrasound,

patient came in our institution with an ultrasound result of a cesarean

scar pregnancy.

1 week prior to admission patient had amenorrhea hence a self-

pregnancy test was done and revealed a positive result, she sought

consult to a private OB and ultrasound was requested. 5 days prior to

admission, ultrasound result is suggestive of a cesarean scar

pregnancy. 3 days prior to admission a repeat scan was done and still

with the same findings hence patient was advised transfer to a tertiary

6
hospital. Due to financial constraints patient opted to sought consult in

our institution and was subsequently admitted.

Her first pregnancy was delivered by a low segment cesarean section

last 2018 in a provincial hospital, indication was unknown.

On physical examination, vital signs were stable, abdominal exam

revealed a midline scar with no mass nor tenderness noted. Speculum

exam the vaginal wall has no lesion, a clean looking cervix with no

erosion, lesion or bleeding noted. On internal exam cervix is firm,

closed, no motion tenderness, uterus small, no adnexal mass nor

tenderness.

A repeat transvaginal ultrasound scan showed a retroverted uterus

with a complex mass located within the myometrium at the anterior

lower uterine segment within the previous cesarean scar measuring

3.20 x 2.53 x 2.89 cm with a gestational sac measuring 1.95 cm by

mean sac diameter containing a yolk sac measures 0.24 cm and an

embryo with crown-rump length of 0.99 cm equivalent to 7 weeks and

1 day age of gestation with fetal heart rate of 143 bpm(Figure 13). A

peritrophoblastic flow demonstrated around the gestational sac on

color Doppler, residual anterior myometrial thickness measures 0.22

cm (Figure 14), the corpus luteum was seen in the right ovary. 3D

scan of the uterus on sagittal view shows a GS with a yolk sac and

embryo at the anterior myometrial wall with an empty endometrial

cavity (Figure 15). No adnexal mass was noted. All laboratory work-up

are within normal limits expect for the serum B-HCG which is elevated

at 37,473 mIU/mL.

7
The patient underwent exploratory laparotomy with ultrasound guided

resection of the caesarean scar pregnancy followed by suction

curettage and repair of the caesarean scar. There was no

hemoperitoneum, uterus is small, previous scar is intact with no

bulging mass noted. Ultrasound was done to identify the exact

location of the gestational sac and to identify the site of incision. A

post evacuation scan was done to confirm the complete removal of

the placental tissues (Figure 16 & 17), the surgery was unremarkable

with an estimated blood loss of 350 cc.

Patient was discharged on postoperative day 2. Histopathologic result

revealed a necrotic decidua and immature placental tissue (Figure

18). On her follow-up patient has no subjective complaint, weekly

serum B-HCG monitoring was done but due to some personal matter

she failed to have her serum B-HCG extracted on the 3 rd week

postoperatively but already revealed normal on the 4 th week (Table 1).

A repeat ultrasound was done after 1 month showing an intact scar

(Figure 19).

8
DISCUSSION:

Cesarean scar ectopic pregnancy is a rare form of ectopic pregnancy,

various theories have been proposed to explain why an embryo would

implant in the LSCS scar. Most describe the blastocyst entering the

myometrium through microscopic tracts from small uterine scar

dehiscences, with the absence of decidua basalis over the scar will

lead to the gestational sac completely embedded in the myometrium,

surrounded by fibrous scar tissues and separate from the endometrial

cavity (2,9).

Lansen and Solomon reported the first patient with CSP in 1978,

since then there is increasing number of reported CSP in literature

from 18 in 2002 to 16 in 2007. The true incidence of CSP is unknown,

however, estimates are that it follows 1:1800 to 1:2500 of all

pregnancies, and 0.15% in women with previous history of cesarean

section which constitutes 6.1 % of ectopic pregnancies in women with

history of at least one previous CS will be CSP (8,11,16). The

increase of cases is attributed to the worldwide increase rate of

cesarean sections and the availability of ultrasonography for the early

pregnancy assessment. There are 2 articles from a locally published

journal reported cases of CSP that are diagnosed at 1 st trimester but

managed differently.

9
 The sole risk factor in developing CSP is a history of previous

pregnancies delivered by cesarean section (7). Few published

literature includes adenomyosis, in vitro fertilization, prior dilatation

and curettage, hysterotomy, myomectomy, abnormal placentation,

manual removal of placenta as additional risk factors in the possible

development of CSP (6,12). Women with more number of cesarean

delivery have higher risk of developing CSP with approximately 72%

of cesarean scar pregnancies occur in women who have had more

than two cesarean deliveries (3). The patient presented had a history

of 2 previous cesarean delivery which made her at risk of developing

CSP.

According to a study by Michener and Dickson of 13 patients with

CSP, vaginal bleeding was the most common presenting symptoms in

9 patients, while in a literature review done by Silver et al of 57

patients with CSP wherein 37% were noted to be asymptomatic, 38%

had painless vaginal bleeding, 16% had painful vaginal bleeding and

9% experienced abdominal pain without vaginal bleeding (2). Our

patient who had vaginal bleeding which is a present in majority of

patient with CSP, and on transvaginal ultrasound revealed an

incidental finding of a cesarean scar pregnancy with that she was

diagnosed and management early that prevented her from developing

life threatening complications.

Ultrasound has advantages of being simple, safe, noninvasive, lower

expense, radiationless and realtime (21).Ultrasonography is the

10
 mainstay is diagnosing CSP, various literature suggest ultrasound

criteria which includes:

a. Empty uterus with clearly visualized endometrium

b. Empty cervical canal

c. Gestational sac implanted in the lower anterior uterine segment at the

presumed site of cesarean section incision site

d. Thin and absent myometrium between the gestational sac and the

bladder (1-3mm)

All of this sonographic criteria in diagnosing CSP are present in our

patients ultrasound findings. This includes an empty endocervical

canal, a gestational sac with yolk sac and embryo with good cardiac

activity that is located in the lower anterior uterine segment above the

internal cervical os and within the previous cesarean scar, empty

uterine cavity and a thin myometrium between the gestational sac and

the bladder measured 0.30 cm.

In addition with this criteria, a prominent peritrophoblastic flow is

demonstrated around the gestational sac on color Doppler (11, 19).

This will help to distinguish CSP from a non-viable detached

intrauterine pregnancy, wherein the gestational sac appears avascular

compared to a viable cesarean scar pregnancy the gestational sac

appears well-perfused on Doppler (19). Another differential diagnosis

to be considered is cervical ectopic pregnancy in which the gestational

sac is implanted in the cervix with the sac located in the endocervical

canal rather than embedded in the anterior lower uterine segment

seen in CSP (17).

11
A three-dimensional (3D) ultrasound can also be used as a diagnostic

tool in diagnosis of CSP as a supplement to 2D ultrasound. Using this

can give us a better visualization of the thin anterior myometrium and

the bladder-uterus surface, which significantly improves the detection

of early placenta accreta which can help in the proper management of

CSP (8,21).

In some cases MRI can be a help in confirming the diagnosis of CSP,

it enables accurate measurement of the distance between the urinary

bladder, myometrium and gestational sac and offers good

visualization of the uterine cavity and cervical canal (16). However,

the use of MRI should be limited to equivocal cases only (9).

However, MRI could not identify the embryo with or without heart

activity. Disadvantages includes high cost, time consuming (21), and it

is not readily available in all institutions in our country.

Vial et al classified CSP into 2 different types. Type I which is also

called endogenic type is when implantation occurs on the scar site

and the gestational sac grows toward the cervico-isthmic or within the

cavity. This give a chance for a live birth, but the risk of massive

bleeding from the implantation side is very high. Type II or the

exogenic type, occurs when the gestational sac is deeply embedded

in the scar and the surrounding myometrium and grows toward the

urinary bladder that leads to uterine rupture and hemorrhage in the

first trimester of gestation (11,16). In relation to the ultrasound done

with our patient we cannot conclude as to what type of CSP it will

progress.

12
In a study by Lin, SY [Link] in 2018 they categorized CSP into 4

grades based on the sonographic findings of JC Shih in 2017,

wherein Grade I CSP when the GS was embedded in less than the

one half thickness of the myometrium. Grade II CSP is when the CSP

occupies more than one half depth of the implanted myometrium.

Grade III CSP, the GS bulged out of the overlying myometrium and

serosa. Grade IV CSP indicated that the GS became an amorphous

tumor with rich vascularity at the site of the previous cesarean scar

(Figure 20). They concluded that this grading revealed an almost

perfect of the reliability for inter-rater agreement on ultrasound grading

(13). Based on this, our patient in this case presented is further

classified as grade II CSP for it occupies more than one half depth of

the implanted myometrium. Using this categorization they suggest

different minimally invasive management approached, TCR

(transcervical resection) can be offered to patients with grade I CSP,

while hysterotomy via LSC or mini-laparotomy for grade II CSP. For

both grade III and IV a hysterotomy via laparotomy or hysterectomy

could be considered, but the risk of bleeding is higher in grade IV CSP

which often requires UAE (uterine artery embolization) (13).

In most cases treatment options should be individualized depending

on the patient’s age, number of previous cesarean deliveries, the

patients hemodynamic condition, desire to preserve fertility and the

available infrastacture and expertise of the clinicians managing her

case (5). There is no consensus on the preferred mode of treatment

with regards to CSP, various cases and series have reported different

13
treatment modalities and options. Therapeutic options include suction

evacuation, systemic or local injection of methotrexate or other

embryocidal agents (KCl), uterine artery embolization. Surgical

treatment which includes wedge resection, hysterotomy, transcervical

resection and hysterectomy.

Deb et. at suggested that medical management using methotrexate

can be considered in patient with CSP if the gestational age is <7

weeks, Beta HCG level of < 5,000 IU/L, mass diameter < 25 mm, no

cardiac activity of the embryo and the presence of the myometrium

between the GS and the bladder wall. However according to Lin SY et

al. with the use of methotrexate or uterine evacuation for a pre-

existing CSP, it may transform into a large vascular tumor that may

lead to catastrophic bleeding often occurs during surgical intervention

(13). This was initially consider as a form of management in this case

but due to ethical issues, possible massive bleeding and the risk of

future recurrent implantation or uterine rupture in the future pregnancy

(20).

The use of ultrasound is not only limited to diagnosing CSP but it can

also be of great importance in its management. A study done by

Bigrardi and Condous wherein 7 out of 1195 women seen in the early

pregnancy unit was diagnosed with CSP, 5 of which were treated with

Transrectal ultrasound-guided surgical evacuation as the primary

treatment and 2 were given methotrexate, one of whom subsequently

underwent TRS-guided aspiration due to failure of conservative

management. They concluded that transrectal ultrasound enable

14
appropriate visualization of the entire uterine cavity and the CSP

which can reduce the possibility of uterine perforation with damage to

surrounding structure (23). Curettage and transcervical resection are

another form of surgical management options for CSP can benefit with

the use of ultrasound guidance as it facilitates complete removal of

placental tissues, shorten the length of the procedure and decrease

the risk of uterine perforation (21,22). Patient who opted for medical

management can also benefit with the ultrasound guided local

injection of methotrexate, it consist of aspiration of the GS and

injection of the methotrexate into the GS. This will give a real-time

supervision of the needle, confirm the scar location and guide a

precise puncture (21).

As for our patients a more conservative surgical management was

done, ultrasound guided wedge resection followed by curettage.

Wedge resection of the CSP was considered by various authors to be

the preferred treatment for women wishing to preserve fertility. This is

because the excision of the old scar not only avoids the possibility of

residual trophoblast being left in situ, but also removes the

microtubular tracts and therefore reduces the risk of recurrence (20).

There are no published article stating the use of ultrasound guidance

in correlation with wedge resection of the CSP, but with the use of

ultrasound we are able to clearly visualize the location and

supervising the puncture path during this surgical procedure which

leads to less blood loss and prevent further surgical complications.

There is no definite guidelines on how should we monitor patient with

15
cs scar pregnancy who underwent surgical management nor medical

management. In a retrospective review done by Ong X and

colleagues, a repeat serum beta HCG was done on post-operative

day 1, then after 2 weeks and 1 week there after and noticed that

there is a decline on the result (25). A 5 years case series done by

Grechukina O et al where in patient who are diagnosed with cs scar

pregnancy who underwent medical management using local or

systemic methotrexate therapy, potassium chloride injection,

hysteroscopy with curettage, uterine artery embolization and using

uterine balloon where monitored by serum beta HCG and ultrasound

evaluations weekly until HCG was undetectable and no mass or

gestational sac was appreciated at the site of the cesarean scar on

transvaginal ultrasound (24). In future pregnancies, early ultrasound

should be used to rule out recurrence, this pregnancies carries

increase risk of morbidly adherent placenta (8). In patient who

underwent surgical repair of a CSP, the time frame required for

adequate healing prior to reattempting pregnancy remains unclear

(26).

16
CONCLUSION:

We presented a rare case of CS scar ectopic pregnancy but due to

the increasing rate of cesarean delivery worldwide, the incidence are

also increasing in number. A thorough history taking and physical

examination on patients with a previous history of cesarean delivery

accompanied by vaginal bleeding with or without abdominal pain, a

high clinical suspicion for a cesarean scar pregnancy in this patient

must be considered.

Ultrasound which is the most accessible, safe, non-invasive, simple

and realtime, hence it is the preferred diagnostic tool in diagnosing

CSP. It can be done transvaginal or in combination with

transabdominal, three-dimensional and color doppler ultrasound,

guided by the suggested ultrasound criteria of a.) empty uterus with

clearly visualized endometrium b.) empty endocervical canal c.)

17
gestational sac implanted in the lower anterior uterine segment at the

presumed site of cesarean section incision site and d.) thin and

absent myometrium between the gestational sac and the bladder (1-3

mm).

This will give us to a more accurate diagnosis and type of CS scar

pregnancy, it’s location, size of the gestational sac and viability of the

embryo if present. Ultrasound is not only use for diagnosing CSP, it

can also be of advantage in the management of CSP may it be

medical or surgical approach. As for the cases presented we utilize

the use of ultrasound thru an ultrasound guided wedge resection of

the CS scar pregnancy (case 1) and in guided surgical incision and

evacuation of the CSP (case 2), both of which were completely

evacuated as confirmed by using ultrasound intra-operatively.

But still an individualized multidisciplinary approach to management is

still needed to prevent future problems, although the risk of future

CSP and uterine rupture is still present.

18
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APPENDICES:

Figure 1. Transvaginal ultrasound image on sagittal view shows the cervix with an
empty endocervical canal. A gestational sac with embryo and yolk sac located

23
 in the anterior lower uterine segment above the internal cervical os with a
dilated endometrial cavity with intracavitary fluid.

EM

Gestaional
sac

Figure 2-3. Three-dimensional ultrasound showing the Gestational sac which is located at the
anterior myometrial wall with intact endometrium.

Figure 4. A Transvaginal scan image shows a gestational sac located anteriorly in the lower
uterine segment with a thin anterior myometrial thickness measuring 0.30 cm as pointed by
the white arrow (Image A) . A Transabdominal scan show and empty endometrium with a
gestational sac and a thin anterior myometrial as pointed by the white arrow (Image B).

24
Figure 5 Pre-operative scan showing the gestational sac with an embryo at the anterior
myometrial wall (blue arrow). The endometrium contains intracavitary fluid ( yellow arrow).

Uterus

Bladder

Figure 6: the bladder (blue arrow) is adherent to the anterior part of the uterus (yellow
arrow)

25
 Figure 7: Thinned anterior lower uterine segment

Figure 8: Post wedge resection of the previous caesarean scar and evacuation
of the products of conception and placental tissues.

26
 A

Figure 9. Specimen labelled (a) the excised CS scar, (b) the Placental tissue and (c) embryo

cervix

EM

Figure 10. showing the uterus post evacuation of the gestational sac and placental tissues

27
Figure 11. On low power view there is a sheet of necrotic decidual tissue infiltrated with
mod. No of inflammatory cells (neutrophils). Noted are few variety of size of chorionic villi.

Figure 12. 1 week post surgery showing an anteverted uterus with intact scar at
the lower uterine segment (yellow arrow ).

28
Figure 13. Transvaginal scan on sagittal view (left) and pelvic scan (right) showing an empty
cervical canal, empty endometrial cavity and a complex mass containing a gestational sac
with a yolk sac and embryo located at the anterior myometrium at the lower uterine
segment within the previous caesarean scar.

A B

Figure 14. Peritrophoblastic flow demonstrated around the gestational sac on color Doppler
(a). the anterior myometrial thickness measures 0.22cm (b).

29
 GS

EM

Figure 15. three dimensional scan showing the complex mass containing a GS is completely
separate from the endometrial cavity.

Figure 16. Post- evacuation of the CS scar pregnancy

30
 Figure 17 Post-evacuation of the Cesarean scar pregnancy (2D gray scale).

Figure 18. On low power view there is a sheet of necrotic decidual tissue traversed by lakes
of hemorrhages and infiltrated by a moderate no. of leukocytic infiltrates. Noted are few
variety of size of chorionic villi.

31
 Figure 19. 2D gray scale scan showing an intact scar (arrow) 1 month post-op

Fig 20. The description of our new ultrasound grading system for CSP.

Grade I CSP represented the depth of CSP embedded in less than one-half thickness of the lower
anterior corpus. Grade II CSP implied CSP occupied more than one-half thickness of the lower anterior
corpus. In grade III CSP, the GS bulged out the overlying myometrium and uterine serosa. In grade IV
CSP, the GS became an amorphous tumor with rich vascularity at the cesarean scar. Adapted from
Lin, SY et al.

TABLE NO.1
CASE 2 Serum B-HCG
32
Admission 37,473.00 mIU/L
Week 1 54.81 mIU/L
Week 2 7.83 mIU/L
Week 3 not done
Week 4 0

33