Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report
Jul 16, 2015
Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report
Case Report
Is there a role for internal iliac artery ligation in post cesareanuterine artery pseudo-aneurysm: A case report
Ahmed S. Elagwany*, Sally S. Eltawab, Ahmed M.F. Mohamed
Department of Obstetrics and Gynecology, Alexandria University, Egypt
a r t i c l e i n f o
Article history:
Received 18 November 2012
Accepted 24 June 2013
Available online xxx
Keywords:
Uterine artery pseudoaneurysm
Caesarian section
Secondary postpartum hemorrhage
Ultrasonography
Computerized tomographic
angiography
Internal iliac artery ligation
a b s t r a c t
Objective: To describe the diagnosis and management of uterine artery pseudoaneurysm
after caesarian section.
Design: Case report.
Setting: Department of Obstetrics and Gynecology.
Patient: A 25-year-old woman developed uterine artery pseudoaneurysm after caesarian
section.
Intervention: Uterine artery pseudoaneurysm after caesarian section was diagnosed on
ultrasonography, computerized tomographic angiography and treated by bilateral internal
iliac artery ligation.
Main outcome measure: Uterine conservation.
Result: Fertility preservation was achieved in the woman.
Conclusion(s): Diagnosis and management of uterine artery pseudoaneurysm after
caesarian section are important to prevent life-threatening hemorrhage caused by pseu-
doaneurysmal rupture.
Copyright ª 2013, Indraprastha Medical Corporation Ltd. All rights reserved.
1. Case report
A 25-year-old para1 female presented to our A&E department
at 6 am in the morning with severe attack of secondary post-
partum hemorrhage. She had uneventful elective cesarean
section six weeks ago in a district hospital due to cephalo-
pelvic disproportion.
On admission, she was very pale tachycardic with heart
rate 124 B/m, BP 80/40 mmHg. Initial resuscitation measures
were done according to our unit protocol with blood samples
were taken for blood tests and cross matching. PV examina-
tion showed a just bulky AVF uterus with severe vaginal
bleeding with blood clots coming through the cervix. Trans-
vaginal U/S showed bulky uterus with endometrial thickness
of 2.5 cm and mild fluid collection in Douglas pouch. Her Hb
was 6 g/dl, platelets count of 210,000/cmm, normal coagula-
tion profile, U&E, liver function.
Examination under anesthesia showed intact vagina and
cervix and profound bleedingwhichwas uterine in originwith
bulky well-contracted uterus. Exploratory laparotomy
through pfannenstiel incision was done which revealed a
perforation of the right lateral uterinewallmeasuring 2� 2 cm
which was covered by clotted blood and necrotic tissues with
heamoperitoneum of about 500 cc. The edges of the defect
were cleaned from blood and necrotic tissue which were
taken for histopathologic examination. 0 vicryl was used to
repair the defect and ensure heamostasis. Peritoneal lavage
was done and intra-peritoneal drain was inserted. The
* Corresponding author. El-shatby Maternity Hospital, Alexandria University, Alexandria, Egypt. Tel.: þ201228254247.E-mail address: [email protected] (A.S. Elagwany).
Available online at www.sciencedirect.com
journal homepage: www.elsevier .com/locate/apme
a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1e4
Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterineartery pseudo-aneurysm: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.06.004
0976-0016/$ e see front matter Copyright ª 2013, Indraprastha Medical Corporation Ltd. All rights reserved.http://dx.doi.org/10.1016/j.apme.2013.06.004
operation lasted for about 45 min during which the patient
received 2 L of fluid, 1.5 L packed RBCs and 3 units of fresh
frozen plasma. The vaginal bleeding stopped and the patient
general condition improved with BP 110/70 mmHg, pulse 90,
urine output about 300, CVP 8 H2O and the patient was then
transferred to the ICU for monitoring, blood transfusion and
follow up. 48 h later the intra-peritoneal drain was removed
and patient transferred to the ward. The histopathology came
back showing only blood and necrotic tissues with no atypia
or malignancy.
The patient improved over the next two days with no
bleeding and her Hb level reached 9.5 g/dl. Unfortunately, on
the third day, the patient experienced severe unprovoked
attack of vaginal bleeding with her Hb level dropped for
5.9 g/dl. Resuscitation measures were initiated and the ultra-
sound examination showed empty uterus with no intra-
peritoneal collection but with hypo-echoic cystic structure
2 cm in diameter attached and related to the right uterine wall
with turbulent flow.
The patient transferred to theater again and under anes-
thesia a 18f Foley’s catheter was inserted intra-uterine and
filled with 30 ml saline for trial of intra-uterine balloon tam-
ponade till reaching final diagnoses which was successful and
the bleeding stopped. The catheter left in place for 48 h during
which correction of the general condition of the patient was
done and blood and plasma transfusionwere taken. After that
48 h the catheter was removed and follow up ultrasound was
donewhichrevealedthatcystic lesion increased indiameterby
1 cm and definite turbulence in Doppler study with initial
diagnosis of arterio-venous malformation (AVM) of the right
uterine artery.
The patient had CT angiography of the pelvis which
showed that a 2 cm pseudoaneurysm is projecting from the
terminal branch of the right uterine artery with a narrow neck
about 2 mm with mild surrounding hematoma (Fig. 1).
The patient was scheduled for embolization two weeks
after. As the patient condition was stable, she opted to be
discharged home with phone contact with emergency
department and strict advice to come back to the hospital if
she feels unwell or vaginal bleeding recurred. Two nights
before the schedules date for embolization, the emergency
department had a phone call from the patient complaining of
a sudden attack of severe vaginal bleeding and she was
advised to come to the hospital immediately.
Twenty minutes later, the patient was in the A&E depart-
ment with an estimated blood loss of about one and half liter.
HerBPwas80/50,HRof 120andHBof 7g/dl andmoderate intra-
abdominal collection on ultrasound. After immediate resus-
citation, patient was transferred to operating theatre after
consenting for laparotomy and hysterectomy. During lapa-
rotomy, the abdomen was filled with blood; the pseudoa-
neurysm was ruptured resulting in a uterine perforation at its
site of about 3 cm in diameter at the same site of previous
perforation. Repair of theuterinedefectwasdoneusing0vicryl
then both internal iliac arteries were double ligated using
0 vicryl. Heamostasis was ensured and intra-peritoneal drain
was inserted for the following 48 h. The procedure took about
90min during which the patient received 2 L of fluid, 3 units of
redbloodcellsand twounitsofplasma.Thepatientadmitted to
the ICU for the next three days then she was discharged to the
ward for another three days. The patient had smooth recovery
with stable general condition and no vaginal bleeding. Trans-
vaginal ultrasound on the fifth day revealed normal size
uteruswithnomasses beside. Pelvis CT angiographywasdone
on the seventh day and come back normal with no aneurysm.
So, the patient discharged home with bi-weekly follow up at
the out-patient gynecology clinic.
The patient general condition improved over the next two
months with no recurrence of the vaginal bleeding and she
was able to breast feed her baby. The patient had a cupper T
380 inserted two months after the operation. She resumed
regularmenses sixmonths after the procedure. One year after
the operation, she had a follow up CT angiography which was
completely normal.
2. Discussion
A pseudoaneurysm is a blood-filled cavity communicating
with the arterial lumen owing to deficiency in one or more
layers of the arterial wall.1 Development of pseudoaneurysms
is a complication of vascular injury resulting from inflam-
mation, trauma, or iatrogenic causes such as surgical pro-
cedures, percutaneous biopsy, or drainage. Pseudoaneurysm
of the uterine artery is a rare but serious complication of gy-
necologic surgery that may be unnoticed in the early post-
operative period. Without precise ultrasonographic and
radiologic diagnosis before the manifestation of symptoms
associated with hemorrhage, these pseudoaneurysms are
prone to unpredictable rupture, resulting in exsanguination
with high morbidity and mortality rates.2
Pseudoaneurysm of the uterine artery is an uncommon
cause of delayed postpartumhemorrhage following caesarean
or vaginal delivery and is potentially life threatening. Typi-
cally, the lesions are discovered because the patients have
symptoms related to delayed rupture of the pseudoaneurysm,
causing hemorrhage.2 A pseudoaneurysm may be asymp-
tomatic, may thrombose, or may lead to distal painful embo-
lization. The risk of rupture is proportional to the size and
intramural pressure. Diagnosis is usually based on both
Doppler sonography and arteriography.3
Occurrence of pseudoaneurysm in the uterine artery is a
rare but serious complication of hysterectomy,2 myomec-
tomy,4 spontaneous vaginal delivery, cesarean section, and
dilatation and curettage. Because the natural history of uter-
ine arterial injury is not well documented and the clinical
appearance of a pseudoaneurysm is variable, precise diag-
nosis of pseudoaneurysm in an asymptomatic patient is
difficult. However, unless recognized before rupture,2 uterine
artery pseudoaneurysm can cause potentially life-threatening
hemorrhage after blood may track through the myometrium
and establish a connection with the uterine cavity.4 With the
introduction of modern imaging modalities, the diagnosis of
uterine artery pseudoaneurysm has become more common,5
allowing early detection and therapeutic intervention before
the pseudoaneurysm manifests clinically, sometimes with
catastrophic results. The ability to diagnose pseudoaneurysm
at an asymptomatic stage is of obvious benefit for all patients
to avoid the potential complications of delayed rupture and
hemorrhage.
a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1e42
Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterineartery pseudo-aneurysm: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.06.004
Regarding diagnostic imaging modalities for pseudoa-
neurysm, the initial usefulness of ultrasonography is well
documented. In general, on grayscale ultrasonography,
pseudoaneurysm has a characteristic sonographic appear-
ance consisting of a pulsating anechoic or hypo-echoic well
defined cystic structure with or without associated pelvic
hematoma or free fluid.4 Color Doppler ultrasonography hel-
ped to establish the diagnosis by demonstrating turbulent
blood flow within the cystic structure. With the advent of
multi-detector row helical CT scanners, three-dimensional CT
angiography is becoming a useful diagnostic modality for
identification of vascular disorders.
Postpartum hemorrhage remains one of the major causes
of maternal mortality. Secondary postpartum hemorrhage is
defined as excessive bleeding starting any time from 24 h
after delivery up to 6 weeks postpartum. Common causes
include retained products of conception, subinvolution of the
placental bed, and endometritis.6 Rare causes include pseu-
doaneurysm of uterine artery and choriocarcinoma. When
the more common causes have been excluded, pelvic angi-
ography may be performed. Uterine artery embolization can
be carried out to control hemorrhage. In 1979, Brown et al
reported the first case of selective arterial embolization used
successfully to treat an extra-uterine pelvic hematoma after
three failed surgical attempts to control the bleeding. Since
then, arterial embolization has been used successfully to
control postpartum bleeding from uterine atony, placenta
accreta, and vulvar and vaginal hematomas. The efficacy and
safety of selective arterial embolization of uterine arteries
was evaluated by Pelage et al in women with delayed sec-
ondary postpartum hemorrhage. In their series of 14 women,
pseudoaneurysms of the uterine artery were found in 2
women.7 Immediate resolution of external bleeding was
observed after embolization. In this series, no complications
related to this invasive treatment were found.7 A true
aneurysm has all three layers of arterial wall, whereas
pseudoaneurysm does not have all the three layers of arterial
wall. The differential diagnosis of pseudoaneurysm includes
acquired arteriovenous malformations (AVMs), arteriove-
nous fistulas, and direct vessel rupture. AVMs are charac-
terized by multiple communications of varying sizes
between arteries and veins, which can be congenital or
acquired.8
Congenital uterine AVMs are due to abnormality in the
embryologic development of primitive vascular structures,
whereas acquired AVM’s consist of multiple small arterio-
venous fistulas between intramural arterial branches and
the myometrial venous plexus. Acquired AVM’s occur more
commonly following D and C, uterine surgery, or trauma to
the uterus. Color flow Doppler demonstrates to-and-fro sign
in the neck of the pseudoaneurysm and yin-yang sign in the
body of the pseudoaneurysm. AVM’s are characterized by
marked aliasing on color flow Doppler and arterialized
venous flow on spectral Doppler evaluation.9
Angiographic embolization has the advantages of
decreased morbidity, ability to localize the bleeding site,
Fig. 1 e CT pelvic scan (a, b) and CT angiography (c, d) showing right uterine pseudo-aneurysm.
a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1e4 3
Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterineartery pseudo-aneurysm: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.06.004
provide a more distal occlusion than surgical ligation and
preservation of future fertility compared to hysterectomy.
Inadequate embolization of a pseudoaneurysm due to extra-
uterine feeding arteries, such as the internal pudendal artery,
ovarian artery, inferior epigastric artery or contralateral
uterine artery may lead to embolization failure.8
In the case of our patient, primary repair of ruptured
pseudo-aneurysm plus bilateral internal iliac ligation was an
effective management for our case. Burchell demonstrated
that bilateral internal iliac artery ligation was more effective
in reducing the pulse pressure than unilateral ligation.9 It is
possible that the redistribution and redirection of blood or
hypoxia-induced neo-vascularization allows bleeding to recur
after unilateral ligation. Hence, bilateral internal iliac artery
ligation is safe and more advantageous than unilateral
ligation.
We conclude that in a woman with unexplained vaginal
bleeding after C-section delivery, pseudoaneurysm is a
potentially life-threatening complication and should be
considered in the differential diagnosis of secondary post-
partum hemorrhage. Although data are scant, bilateral inter-
nal iliac artery ligation for obstetric hemorrhage appears to
have no increased deleterious effect on future fertility and is
more effective when compared to unilateral ligation.
Conflicts of interest
All authors have none to declare.
r e f e r e n c e s
1. Zimon AE, Hwang JK, Principe DL, Bahado-Singh RO.Pseudoaneurysm of the uterine artery. Obstet Gynecol. 1999;94:827e830.
2. Langer JE, Cope C. Ultrasonographic diagnosis of uterine arterypseudoaneurysm after hysterectomy. J Ultrasound Med. 1999;18:711e714.
3. Hidar S, Bibi M, Atallah R, Essakly K, Bouzakoura C, Hidar M.Pseudoaneurysm of the uterine artery: Apropos of 1 case. JGynecol Obstet Biol Reprod (Paris). 2000;29:621e624.
4. Sizzi O, Rossetti A, Malzoni M, et al. Italian multicenter studyon complications of laparoscopic myomectomy. J MinimInvasive Gynecol. 2007;14:453e462.
5. McGonegle SJ, Dziedzic TS, Thomas J, Hertzberg BS.Pseudoaneurysm of the uterine artery after an uncomplicatedspontaneous vaginal delivery. J Ultrasound Med. 2006;25:1593e1597.
6. Khong TY, Khong TK. Delayed postpartum hemorrhage: amorphologic study of causes and their relation to otherpregnancy disorders. Obstet Gynecol. 1993;82:17e22.
7. Brown BJ, Heaston DK, Poulson AM, Gabertet HA, Mineau DE,Miller Jr FJ. Uncontrollable postpartum bleeding: a newapproach to hemostasis through angiographic arterialembolization. Obstet Gynecol. 1979;54:361e365.
8. Kwon JH, Kim GS. Obstetric iatrogenic arterial injuries of theuterus: diagnosis with US and treatment with transcatheterarterial embolization. Radiographics. 2002;221:35e46.
9. Kovo M, Behar DJ, Friedman V, Malinger G. Pelvic arterialpseudoaneurysm e a rare complication of cesarean section:diagnosis and novel treatment. Ultrasound Obstet Gynecol.2007;30:783e785.
a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1e44
Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterineartery pseudo-aneurysm: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.06.004
Apollo hospitals: http://www.apollohospitals.com/Twitter: https://twitter.com/HospitalsApolloYoutube: http://www.youtube.com/apollohospitalsindiaFacebook: http://www.facebook.com/TheApolloHospitalsSlideshare: http://www.slideshare.net/Apollo_HospitalsLinkedin: http://www.linkedin.com/company/apollo-hospitalsBlog:Blog: http://www.letstalkhealth.in/