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: Ahmedsamyagwany@gmail.com (A.S. Elagwany).

Available online at www.sciencedirect.com

journal homepage: www.elsevier .com/locate/apme

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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.

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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.

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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.

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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

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