Background: Cesarean scar ectopic pregnancies are rare, with an incidence of approximately 1 in 2,000 pregnancies. The trauma of a cesarean section and a subsequent cesarean scar pregnancy can lead to the formation of an arteriovenous malformation (AVM). The resulting intractable bleeding is difficult to manage and can result in an emergent surgical intervention that could jeopardize a female's ability to become pregnant in the future

Gala, Rajiv B.

Smith, Evan S.

University of Queensland eSpace

Successful Management of Cesarean Scar Pregnancy
Complicated by an Arteriovenous Malformation
Evan S. Smith, MD,1 Rajiv B. Gala, MD1,2
1Department of Obstetrics and Gynecology, Ochsner Clinic Foundation, New Orleans, LA 2The University of Queensland School of Medicine,
Ochsner Clinical School, New Orleans, LA
Background: Cesarean scar ectopic pregnancies are rare, with an incidence of approximately 1 in 2,000 pregnancies. The
trauma of a cesarean section and a subsequent cesarean scar pregnancy can lead to the formation of an arteriovenous
malformation (AVM). The resulting intractable bleeding is difficult to manage and can result in an emergent surgical
intervention that could jeopardize a female’s ability to become pregnant in the future.
Case Report: A 29-year-old female, gravida 2 para 1, with 1 prior low transverse cesarean section had a presumed cervical
ectopic pregnancy treated with intramuscular methotrexate. Despite a negative serum beta human chorionic gonadotropin
result, she had a persistent mass in the lower uterine segment of her cesarean section scar and was finally diagnosed with an
AVM. We successfully preserved her fertility by performing a wedge resection of the AVM and using a novel technique of
bilateral O’Leary sutures to occlude the ascending and descending branches of the uterine artery along with intramuscular
vasopressin infiltration.
Conclusion: A multidisciplinary approach involving obstetrics/gynecology, interventional radiology, and anesthesiology
allowed for a safe conservative surgical approach and the preservation of our patient’s fertility.
Keywords: Arteriovenous malformations, cesarean section, pregnancy–ectopic
Address correspondence to Rajiv B. Gala, MD, Department of Obstetrics and Gynecology, Ochsner Clinic Foundation, 4429 Clara St., Suite
440, New Orleans, LA 70115. Tel: (504) 842-9618. Email: rgala@ochsner.org
INTRODUCTION
Ectopic pregnancies complicate 1%-2% of all first-
trimester pregnancies in the United States but still account
for up to 6% of pregnancy-related deaths.1 While 95% of
ectopic pregnancies implant in the fallopian tube, the
remaining 5% are found elsewhere, including the ovary,
peritoneal cavity, cervix, and cesarean section scar.
Arteriovenous malformations (AVMs) of the uterus are
rare, and the incidence is unknown.2 They can be
congenital, thought to be the result of abnormal embryo-
logic differentiation, or acquired. The limited literature
addressing AVMs of the uterus notes that acquired lesions
usually occur after trauma or uterine instrumentation.3
Treatment of nontubal ectopic pregnancies can be
perplexing because of the variety of anatomic implantation
sites and associated implications. This report focuses on
one such case and offers insight into management options
for a patient with a rare, potentially fatal malformation who
strongly desired fertility preservation.
CASE REPORT
A 29-year-old, gravida 2 para 1 patient with a history
significant for 1 previous low transverse cesarean section
presented to an outside hospital with a concern of
persistent, light vaginal bleeding. Her serum beta human
chorionic gonadotropin (hCG) level was 2,500 mIU/mL. A
nonviable fetus measuring 7 weeks, 5 days of gestation by
crown-rump length was noted very low in the uterus and
thought likely to be in the cervix. Differential diagnoses
included cervical pregnancy vs incomplete spontaneous
abortion, and the patient was followed up with repeat
ultrasound the next day. The follow-up ultrasound was
unchanged, and the presumed diagnosis was a cervical
ectopic pregnancy. The patient was treated with 1 dose of
intramuscular methotrexate and responded appropriately,
with a gradual serum beta hCG level reduction to <10
mIU/mL; however, repeat ultrasounds 1 and 2 months after
the original diagnosis noted a persistent 4.8 3 4.9 cm
cervical pregnancy despite the low beta hCG level. The
patient was referred to Ochsner Clinic Foundation for further
management.
At the time of presentation, the patient reported 1 episode
of heavy vaginal bleeding after intercourse the day prior and
ongoing scant bleeding. During physical examination, a
small amount of old blood was observed in the vagina, with
no cervical lesions, cervical abnormalities, or active vaginal
bleeding. Her hemoglobin and hematocrit values were 10.7
g/dL and 32.6%, respectively. Her beta hCG level was 3.5
268 The Ochsner Journal
The Ochsner Journal 15:268–271, 2015
 Academic Division of Ochsner Clinic Foundation
CASE REPORT
mIU/mL, and an ultrasound performed by a maternal fetal
medicine physician showed a large mass (6.03 4.6 cm) in
the lower uterine segment/upper portion of the cervix that
appeared to protrude from the lower uterine segment in the
area of her cesarean section scar, most consistent with a
cesarean scar pregnancy. In comparison with images
obtained 2 weeks prior, the gestational sac previously
visualized was now smaller and more irregularly shaped,
consistent with the patient’s report of a large amount of
vaginal bleeding. Large areas of blood flow were noted,
predominantly on the periphery of the mass (Figure 1).
Differential diagnosis was expanded to include cesarean
scar ectopic pregnancy in addition to the referring physi-
cian’s diagnosis of cervical ectopic pregnancy. The patient
strongly desired fertility preservation and opted for treat-
ment via selective embolization of the pregnancy’s blood
supply.
The interventional radiology department was consulted for
uterine artery embolization. During the attempt at selective
embolization, the femoral artery approach arteriogram re-
vealed rapid venous outflow from the hypervascular uterine
lesion into either the right ovarian vein, internal iliac vein to the
inferior vena cava, or dilated lumbar veins. Because of
concern for an AVM or arteriovenous fistula, the procedure
was aborted, and magnetic resonance imaging (MRI) was
recommended for further evaluation. The MRI was significant
for a large area of heterogeneity involving the anterior lower
uterine segment and a thinned anterior uterine wall with
abnormal, large serpiginous vessels confined by the uterine
wall, possibly in the cesarean scar, with findings related to an
AVM or arteriovenous fistula (Figure 2). After confirmation,
embolization was deemed inappropriate, and the treatment
options offered to the patient were wedge resection of the
lower uterine segment or hysterectomy. The patient strongly
desired uterine preservation and chose to proceed with a
uterine wedge resection.
The patient presented to the emergency department the
evening prior to the planned surgery with reported heavy
vaginal bleeding, weakness, and dizziness. Clotted blood in
the vagina without any brisk bleeding was observed during
physical examination. Hemoglobin and hematocrit values
were significantly decreased to 6.9 g/dL and 21.7%,
respectively. The patient was admitted and transfused with
2 units of packed red blood cells with plans to proceed with
surgery in the morning as originally scheduled.
The patient underwent an exploratory laparotomy with a
wedge resection of the lower uterine segment. A central
venous catheter was placed prior to the start of the
operation in case of the need for rapid fluid resuscitation.
Pfannenstiel incision was used to enter the abdominal
cavity, and findings included a 6-week sized, mobile uterus
with a 334 cm AVM in the lower uterine segment (Figure 3).
Dense bladder adhesions to the lower uterine segment
were dissected to clearly isolate the AVM. Next, the uterine
arteries were ligated bilaterally with O’Leary sutures.
Figure 1. Ultrasound shows large areas of blood flow,
predominantly on the periphery of the large mass (6.034.6
cm) in the lower uterine segment in the area of the patient’s
cesarean section scar.
Figure 2. Magnetic resonance angiogram demonstrates
multiple vessels tangled in the lower pelvis arising from
the right internal iliac artery, corresponding to the arterio-
venous malformation or arteriovenous fistula seen on
magnetic resonance imaging.
Figure 3. A 334 cm arteriovenous malformation is visible in
the lower uterine segment.
Smith, ES
Volume 15, Number 3, Fall 2015 269
Vasopressin was injected around the mass, and a No. 11
scalpel blade was used to score the uterus in an elliptic
fashion around the AVM, with this incision carried posteri-
orly into the endometrial cavity. A significant amount of
bright red, freshly clotted blood was present within the
uterus, indicative of bleeding during the surgery, and the
patient received an additional 3 units of packed red blood
cells and 2 units of fresh frozen plasma intraoperatively. A 2-
layer closure was used for the uterine incision with deep 0-
chromic simple interrupted sutures and 0-chromic running
locked sutures for the superficial layer. Hemostasis was
noted prior to closing the abdominal incision, and the total
estimated blood loss during the procedure was 400 mL.
Cystoscopy at the end of the case did not suggest any
ureteral or bladder injury. Surgical pathology confirmed a
partially thrombosed AVM with dense fibrosis and scattered
chronic inflammation without any products of conception.
The patient’s postoperative course was uncomplicated.
Vaginal bleeding decreased significantly, and postoperative
hemoglobin/hematocrit levels were stable. She was dis-
charged on postoperative day 2 in stable condition. In
addition to bleeding and pelvic rest precautions, the patient
was advised to avoid pregnancy for at least 6 months to allow
the lower uterine segment time to heal, and she was also
made aware of the need for a cesarean section for future
pregnancies. For contraception, she decided to use an
intrauterine device that was successfully placed several
weeks after her surgery. To date, we are unaware of
subsequent pregnancies or pregnancy outcomes for this
patient.
DISCUSSION
Of the few reported cases of uterine AVMs, acquired
occurrences are more common than congenital incidenc-
es.4 AVMs usually follow uterine trauma, such as cesarean
section, pelvic surgery, or uterine curettage, and form
during tissue healing when an artery and veins connect
abnormally. Infection, retained products of conception,
gestational trophoblastic disease, gynecologic malignan-
cies, and exposure to diethylstilbestrol are additional
identified risk factors for acquired uterine AVMs.4
The presenting scenario of uterine AVM is often intractable
bleeding during pregnancy or after uterine trauma. Ultra-
sound, computed tomography, and MRI are frequently used
to evaluate bleeding and suspected AVMs, but angiography
is the gold standard for diagnosis.5 The treatment of choice
for postmenopausal patients is hysterectomy; however,
stable patients who wish to preserve fertility have options.4
Expectant management and/or medical management with
oral contraceptive pills or methylergonovine maleate have
been described as successful options, as have invasive
options such as arterial embolization, hysteroscopy AVM
coagulation, laparoscopic uterine vessel cauterization, surgi-
cal uterine artery ligation, and AVM excision.2
While cesarean scar pregnancy is observed more often
than uterine AVM, it is still a rare occurrence. The reported
incidence of cesarean scar pregnancies is on the rise,
accompanying increased cesarean section rates, with a
2007 report concluding that approximately 1 in 2,000
pregnancies is complicated by cesarean scar implanta-
tions.6 Presentation is most common between 5 and 16
weeks of gestation, with the most common chief concern
being painless light vaginal bleeding. Ultrasound is the first-
line imaging modality for diagnosis in the hemodynamically
stable patient, with color flow Doppler ultrasound, MRI,
diagnostic hysteroscopy, and diagnostic laparoscopy pos-
sibly useful adjuncts for diagnosis and treatment planning.
Treatment should include measures to prevent rupture of
the sac, to remove the gestational sac, and to preserve
fertility. Treatment options are numerous and include
expectant management (although not advised); conserva-
tive medical treatment (methotrexate only); combined
medical treatment (local injection of potassium chloride,
methotrexate, and mifepristone); and medical treatment
combined with cesarean surgical sac aspiration, uterine
curettage, hysteroscopic evacuation, laparoscopic removal,
open surgical treatment, or hysterectomy.6
The incidence of cesarean scar pregnancy complicated
by uterine AVM is unknown, and we found only 5 case
reports to date.7-10 All treatments described in prior cases
included uterine artery or selective arterial embolization.
Methotrexate, uterine curettage, and wedge resection are
also described as successful adjuvant treatment methods.
The AVM in this patient could not be treated with selective
embolization, prompting the decision for surgical manage-
ment. To control bleeding during the wedge resection of the
uterus, O’Leary sutures, traditionally used at the time of
cesarean section for management of uterine artery lacera-
tions from hysterotomy extensions, were placed on the
ascending and descending uterine arteries above and below
the lateral edges of the AVM (Figure 4). This suture placement
helped decrease pulse pressure and allowed for safe excision
and repair. Additionally, there were no long-term conse-
quences to the uterus from the O’Leary sutures because of
good collateral blood supply and the nonpermanent nature of
the suture. The uterus was closed in a multilayer fashion to
develop a strong lower uterine segment that would not
rupture with subsequent pregnancies. The patient was
counseled on the importance of spacing the pregnancy
interval after uterine repair to allow adequate healing.
Figure 4. Bilateral O’Leary sutures, traditionally used at the
time of cesarean section for management of uterine artery
lacerations from hysterotomy extensions, were placed on
the ascending and descending uterine arteries above and
below the lateral edges of the arteriovenous malformation.
Cesarean Scar Pregnancy with Arteriovenous Malformation
270 The Ochsner Journal
CONCLUSION
This case is unique because of the rare incidence of
cesarean scar pregnancy and AVM complication and
because the treatment was performed without prior
uterine artery or selective embolization, unlike the cases
presented in the literature. Our case shows that safe,
effective surgical management can be provided and the
uterus can be preserved for a patient with a potentially
morbid uterine AVM at the cesarean scar site. With the
advent of improved suturing techniques to ensure a
double-layer closure of the lower uterine segment
available in the laparoscopic and robotic platforms, a
minimally invasive approach using our technique could
be considered in the future. Also, a multidisciplinary
approach involving obstetrics/gynecology, interventional
radiology, and anesthesiology allowed for a safe conser-
vative surgical approach.
ACKNOWLEDGMENTS
The authors have no financial or proprietary interest in the
subject matter of this article.
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This article meets the Accreditation Council for Graduate Medical Education and the American Board of Medical
Specialties Maintenance of Certification competencies for Patient Care and Medical Knowledge.
Smith, ES
Volume 15, Number 3, Fall 2015 271


Successful management of cesarean scar pregnancy complicated by an arteriovenous malformation

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