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 cesarean
uterine artery pseudo-aneurysm: A case rep...
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
fr...
Regarding diagnostic imaging modalities for pseudoa-
neurysm, the initial usefulness of ultrasonography is well
documented...
provide a more distal occlusion than surgical ligation and
preservation of future fertility compared to hysterectomy.
Inad...
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Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report

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A pseudoaneurysm is a blood-filled cavity communicating with the arterial lumen owing to deficiency in one or more layers of the arterial wall. Development of pseudoaneurysms is a complication of vascular injury resulting from inflammation, trauma, or iatrogenic causes such as surgical procedures, percutaneous biopsy, or drainage. Pseudoaneurysm of the uterine artery is a rare but serious complication of gynecologic 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.

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Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report

  1. 1. Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report
  2. 2. Case Report Is there a role for internal iliac artery ligation in post cesarean uterine 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 bleeding which was uterine in origin with bulky well-contracted uterus. Exploratory laparotomy through pfannenstiel incision was done which revealed a perforation of the right lateral uterine wall measuring 2 Â 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 a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1 e4 Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterine artery 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
  3. 3. 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 transfusion were taken. After that 48 h the catheter was removed and follow up ultrasound was donewhichrevealedthatcysticlesionincreasedin diameter by 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. Her BP was 80/50, HR of 120 and HB of 7 g/dl and moderate 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 the uterine defect was done using 0 vicryl 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 90 min during which the patient received 2 L of fluid, 3 units of redbloodcells andtwounitsof plasma.Thepatient admitted 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 uterus with no masses beside. Pelvis CT angiography was done 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 regular menses six months 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 postpartum hemorrhage 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 ) 1 e42 Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.06.004
  4. 4. 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 ) 1 e4 3 Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.06.004
  5. 5. 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 artery pseudoaneurysm 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. J Gynecol Obstet Biol Reprod (Paris). 2000;29:621e624. 4. Sizzi O, Rossetti A, Malzoni M, et al. Italian multicenter study on complications of laparoscopic myomectomy. J Minim Invasive Gynecol. 2007;14:453e462. 5. McGonegle SJ, Dziedzic TS, Thomas J, Hertzberg BS. Pseudoaneurysm of the uterine artery after an uncomplicated spontaneous vaginal delivery. J Ultrasound Med. 2006;25: 1593e1597. 6. Khong TY, Khong TK. Delayed postpartum hemorrhage: a morphologic study of causes and their relation to other pregnancy disorders. Obstet Gynecol. 1993;82:17e22. 7. Brown BJ, Heaston DK, Poulson AM, Gabertet HA, Mineau DE, Miller Jr FJ. Uncontrollable postpartum bleeding: a new approach to hemostasis through angiographic arterial embolization. Obstet Gynecol. 1979;54:361e365. 8. Kwon JH, Kim GS. Obstetric iatrogenic arterial injuries of the uterus: diagnosis with US and treatment with transcatheter arterial embolization. Radiographics. 2002;221:35e46. 9. Kovo M, Behar DJ, Friedman V, Malinger G. Pelvic arterial pseudoaneurysm 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 ) 1 e44 Please cite this article in press as: Elagwany AS, et al., Is there a role for internal iliac artery ligation in post cesarean uterine artery pseudo-aneurysm: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.06.004
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