Post-cesarean section pseudoaneurysm of uterine artery: A case report.

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This case report describes a 25-year-old woman who developed three episodes of per vaginal bleeding at 15, 24, and 34 days after an elective lower segment cesarean section, with progressive anemia and no other infectious or bleeding-disorder indicators. Using Doppler ultrasonography, a vascular channel was suggested as a pseudoaneurysm, and CT angiography confirmed a left uterine artery pseudoaneurysm at the cesarean scar site; the limitation is that it is a single-patient report with no comparison group. She was stabilized with transfusion and managed with uterine artery embolization using PVA particles for the distal portion and a platinum coil at the aneurysm neck, after which angiography showed non-opacification and symptoms resolved with obliteration on follow-up Doppler. Relevance to endometriosis: the article briefly mentions that uterine artery pseudoaneurysm can occur after laparoscopic excision of deep endometriotic lesions, though its main focus is secondary postpartum hemorrhage from a cesarean-associated uterine artery pseudoaneurysm rather than endometriosis or adenomyosis.

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Abstract

Introduction and importanceUterine artery pseudoaneurysm is an extraluminal collection of blood caused by damage to the arterial wall which can result from cesarean section, myomectomy, hysterectomy, laparoscopic excision of deep endometriotic lesions, dilation and curettage and uterine cervical conization. Uterine artery pseudoaneurysm may go unnoticed as a possible cause of post-partum hemorrhage.Case presentationWe report a case of 25 years female who presented with 3 episodes of per vaginal bleeding on the 34th postoperative day of cesarean section. Computed tomography angiogram confirmed the aneurysm of the left uterine artery and the artery was embolized for the management of aneurysm. Follow-up scan showed normal blood flow and her symptoms were relieved after the procedure.DiscussionUterine artery pseudoaneurysm can be diagnosed by Doppler ultrasound which shows intrauterine mass with swirling blood flow. The gold standard investigation is computed tomography angiography. Uterine artery embolization is the recent treatment approach which is both safe and effective.ConclusionWomen who present with postpartum vaginal bleeding should be promptly evaluated for uterine artery pseudoaneurysm. Uterine artery pseudoaneurysm can then be effectively managed through embolization, ensuring timely intervention and improved maternal health outcomes.
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Case

A 25-year-old female presented on the 34th postoperative day of elective Lower Segment Cesarean Section (LSCS) with a complaint of three episodes of per vaginal bleeding. She had the first episode of per vaginal bleeding after 15 days, the second episode after 24 days, and the third episode 34 days after LSCS. For each episode of bleeding, she used 4–5 pads per day which were fully soaked with the passage of clots. It was associated with dizziness and generalized weakness of the body. She was a P3L3 (parity three living three) with normal delivery seven years back and emergency LSCS three years back. There were no maternal and fetal complications in her previous two pregnancies. She had no history of dysuria, burning micturition, urgency, trauma, pain abdomen, per vaginal discharge, fever, chest pain and shortness of breath. There were no similar episodes in the past. There was no family history of bleeding disorders. She did not smoke, consume alcohol or took any medications. At the presentation, she appeared pale, her vitals were stable and the rest of the examinations were found to be within physiological limits. Per speculum examination showed cervix was smeared with blood but no active bleeding and per vaginal examination revealed an anteverted uterus of eight weeks size, mobile, non-tender, all fornices were free, os closed, and with no cervical motion tenderness. During the first episode, the patient visited a tertiary hospital near her residency. Hemoglobin was 13.3 g/dL and USG findings of abdomen and pelvis were normal. She was admitted, given ceftriaxone and tranexamic acid and after observation for two days, her symptoms resolved and she was discharged with the same medications. During her second episode, hemoglobin was 9.4 g/dL and USG showed a well defined smooth thin walled avascular structure 9.3 ∗ 7.6 ∗ 10 mm at myometrium just anterior to endometrial at the cesarean scar site suggestive of retention myometrial cyst or hematometra. She was admitted again and managed conservatively with the same medications and discharged after two days. During her third episode, she came to our center for a second opinion about her recurrent per vaginal bleeding. Several investigations were performed on the day of presentation: hemoglobin, complete blood count, prothrombin time-international normalized ratio (PT-INR), random blood sugar, renal function test, urine pregnancy test, urine routine and microscopic examination, and USG of abdomen and pelvis ( Fig. 1 ). Hemoglobin was 7.5 g/dL, hematocrit 22 %, total red blood cell (RBC) count 2.5 million/microL, PT 15.6 s, INR 1.2, total leukocyte count 11.9 thousand/microL, urine pregnancy test was negative and routine and microscopic examination of urine was normal. Fig. 1 USG showing uterus of size 8.4 ∗ 4 ∗ 4.8 cm size and well-defined cystic lesion (black arrow) in lower uterine cavity measuring 11 ∗ 8 mm. Fig. 1 USG showing uterus of size 8.4 ∗ 4 ∗ 4.8 cm size and well-defined cystic lesion (black arrow) in lower uterine cavity measuring 11 ∗ 8 mm. On the first day of hospital admission peripheral blood smear (PBS), reticulocyte percentage, iron profile, blood group, and cross-match were sent. Normocytic normochromic to microcytic hypochromic RBC were seen in PBS. The reticulocyte percentage was 4.4 % and iron profile report was normal. Doppler USG showed a prominent anechoic vascular channel adjacent to the posterior myometrium giving the impression of pseudoaneurysm ( Fig. 2 ). Fig. 2 Transvaginal duplex Doppler sonogram of the aneurysm showing high velocity, high resistance arterial flow within it. Fig. 2 Transvaginal duplex Doppler sonogram of the aneurysm showing high velocity, high resistance arterial flow within it. The patient was transfused 2 pints of whole blood. After the patient was hemodynamically stable, we sent the patient for a CT angiogram which suggested left uterine artery pseudoaneurysm ( Fig. 3 , Fig. 4 ). Fig. 3 CT Angiogram showing well defined focal outpouching (orange arrow) of 1.5 ∗ 1.5 ∗ 1.3 mm from the branch of the left uterine artery within anterior myometrium at cesarean section site. Fig. 3 Fig. 4 Volume rendered multidetector computed tomography image showing a pseudoaneurysm in the left uterine artery (black arrow). Fig. 4 CT Angiogram showing well defined focal outpouching (orange arrow) of 1.5 ∗ 1.5 ∗ 1.3 mm from the branch of the left uterine artery within anterior myometrium at cesarean section site. Volume rendered multidetector computed tomography image showing a pseudoaneurysm in the left uterine artery (black arrow). We decided that embolization of the left uterine artery would be an effective management option for the patient and the case was handed over to the intervention radiology team. Following all aseptic precautions, local anesthesia was administered and the right common femoral artery was punctured under USG guidance and confirmed with fluoroscopy. The contralateral iliac artery was cannulated and an angiogram was done using 4 Fr cobra catheter ( Fig. 5 ). A Progreat microcatheter 2.4 Fr was used for superseletive cannulation of the left uterine artery. Polyvinyl Alcohol (PVA) 300–500 μm was used to embolise the distal portion and terumo platinum coil (3 mm diameter, 20 mm length) was used to embolise the proximal door of the aneurysm. Fig. 5 Pre embolization selective arteriogram demostrating focal outpouching of the vessel in the distal left uterine artery (blue arrow). (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.) Fig. 5 Pre embolization selective arteriogram demostrating focal outpouching of the vessel in the distal left uterine artery (blue arrow). (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.) Post-embolization angiogram showed non-opacification of the aneurysm which suggested omission of the aneurysm from the circulation ( Fig. 6 ). The rest of the vessels were normal in the angiogram. The procedure was uneventful. The patient was hemodynamically stable throughout the procedure. Manual compression of the femoral artery was used to achieve hemostasis at the puncture site after the procedure. Fig. 6 Postembolization, selective arteriogram of the left internal iliac artery showing placement of metallic coils (orange arrow) at the orifice of the pseudoaneurysm within the left uterine artery. Fig. 6 Postembolization, selective arteriogram of the left internal iliac artery showing placement of metallic coils (orange arrow) at the orifice of the pseudoaneurysm within the left uterine artery. A review scan was performed on the third post procedure day and the patient was discharged ( Fig. 7 ). The post-procedure recovery was good and her symptoms were relieved after the procedure. Fig. 7 Doppler USG showing obliteration of the pulsatile cystic lesion. Fig. 7 Doppler USG showing obliteration of the pulsatile cystic lesion.

Author

Poudel A, Thapa A, Giri A, Paudel A, Sah A and Luitel S designed the report, reviewed the literature, edited the images, and wrote the paper; Thapa S revised the paper. All the authors have read and approved the final manuscript.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Ethical

Based on our institutional policy, ethical approval is not required for case reports.

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Research

N/A.

Guarantor

Sabita Thapa.

Disclaimer

No patient or author details are included in the figures.

Discussion

We reported a case of a 25-year-old female who presented with 3 episodes of per vaginal bleeding 32 days following LSCS. After a detailed history, examination, and investigations, it was found that the symptoms had developed due to a rupture of the left uterine artery pseudoaneurysm. When a punctured or lacerated artery does not seal completely, blood escapes, dissects the adjacent tissues, and collects in perivascular areas. If this collection maintains communication with the parent vessel, a pseudoaneurysm can result. Pseudoaneurysms have a neck that typically does not close spontaneously once past a certain size. The blood in the pseudoaneurysm is contained by fibrin/platelet crosslinks [ 1 ]. Pseudoaneurysms also known as false aneurysms, are complications of vascular injury. Pseudoaneurysm of the uterine artery can result from injury during cesarean section, myomectomy, hysterectomy, laparoscopic excision of deep endometriotic lesions, dilation and curettage and uterine cervical conization [ [4] , [5] , [6] , [7] , [8] , [9] , [10] ]. The boundaries of a pseudoaneurysm are formed by a peripheral thrombus and are not surrounded by three arterial layers as in true aneurysms [ 12 ]. In our case, the pseudoaneurysm might have resulted from the cesarean section. The American College of Obstetricians and Gynecologists defines secondary PPH as bleeding 24 h to 12 weeks after delivery [ 13 ]. Secondary PPH is commonly caused by retained products of conception (RPOC), subinvolution of the placental bed, and infection. Less common etiologies include bleeding disorders, arterial pseudoaneurysms, arteriovenous fistulas, choriocarcinoma, cervical carcinoma, adenomyosis, infected polyps or fibroids, uterine diverticulum, excessive bleeding with menses resumption, hypoestrogenism, and cesarean scar dehiscence [ 2 ]. A study done in Thailand found the incidence of secondary PPH to be 0.21 % [ 14 ]. UAP might present with primary as well as secondary PPH. The prevalence of UAP was 0.51 % among patients with primary PPH and 2.55 % among secondary PPH [ 3 ]. UAP can be diagnosed based on color Doppler USG which shows intrauterine mass with swirling blood flow, with a to-and-fro or yin-and-yang pattern. Magnetic resonance imaging (MRI) can be used which reveals an enhanced, pseudoaneurysmal sac-like structure within the uterus. The gold standard investigation is CT angiography confirming the presence of UAP with a narrow connection with the parent uterine artery [ 12 ]. In our case, Doppler USG raised the suspicion of pseudoaneurysm which was confirmed by a CT angiography. Open surgical management, including hysterectomy and ligation of uterine or internal iliac artery used to be the treatment of choice for UAP in the past. However, the surgical management is reserved for larger pseudoaneurysms, pseudoaneurysms without a narrow neck, and those with failed compression or thrombin injection [ 15 ]. Uterine balloon tamponade and laparoscopic surgery have also been used for the treatment of UAP [ 16 , 17 ]. Recent modality of treatment has shifted to Uterine Artery Embolization (UAE), which is safe and effective [ 12 ]. Therapeutic embolization is defined as the deliberate introduction of occluding material into a blood vessel in order to reduce or obstruct blood flow. The choice of embolic agent for a specific application depends on the degree of permanence and the desired level of occlusion. Temporary embolic agents like gel foam and permanent embolic agents such as coils, vascular plugs, detachable balloons, PVA particulates, glue, alcohol, and sclerosants are used for sustained occlusion in interventional procedures. Coils are made from either stainless steel or platinum and have Dacron fibers placed at right angles to the long axis of the coil to increase the surface area and thereby increase the speed and permanence of thrombosis. Coils rely on mechanical obstruction, platelet activation, and the patient's own clotting cascade to fully occlude vessels. PVA particles produce mechanical occlusion of the vessel in addition to activation of thrombin and inducing fibroblast ingrowth, which leads to a relatively permanent occlusion [ 18 ]. UAE has a special benefit for patients who desire fertility preservation [ 19 ]. In our case, UAE was the treatment of choice. Some studies have shown the spontaneous resolution of UAP [ 20 , 21 ]. There are a few case reports of a pseudoaneurysm of the uterine artery after cesarean section. Shahab et al. reported a case of a 29-year-old woman who underwent a cesarean section and IUD placement and was found to have a pseudoaneurysm of 1.6 cm in the left uterine artery on a follow-up USG. CT angiography was done for confirmation and uterine artery embolization was done [ 10 ]. Another case was reported by Pandey et al. in 2023. A 31-year-old woman presented with refractory secondary postpartum hemorrhage after 44 days of cesarean section. The patient was in shock and found to have a ruptured UAP in CT angiography. Emergency trans-arterial UAE was performed, aiding in the stabilization and recovery of the patient [ 22 ]. In 2021, Gupta et al. reported a case of a 27-year-old female who underwent a cesarean section and presented with two episodes of vaginal bleeding three weeks after delivery. Transvaginal sonography (TVS) with color Doppler USG was done which showed pseudoaneurysm which was confirmed by CT angiogram. Bilateral uterine arteries were embolised using gel foam to treat her [ 23 ]. A study done by Feld et al. reported a case of a 37-year-old female presenting with bleeding during pregnancy due to UAP which was confirmed in the postpartum period [ 24 ]. Women who present with vaginal bleeding during the post-partum period should undergo a thorough evaluation for UAP. While UAP is relatively uncommon, its diagnosis can be effectively confirmed through the use of CT Angiography. Once diagnosed, UAP can be appropriately managed through embolization procedures. This systematic approach ensures timely identification and intervention for this potentially serious postpartum complication, contributing to improved maternal health outcomes.

Provenance

Not commissioned, externally peer-reviewed.

Introduction

A pseudoaneurysm is an extra-luminal collection of blood caused by damage to the arterial wall, resulting in locally contained hematoma with turbulent blood flow [ 1 ]. Uterine artery pseudoaneurysms (UAP) are rare causes of postpartum hemorrhage (PPH) with a prevalence of 5–6 in 1000 cases of PPH [ 2 , 3 ]. They may result from laceration or injury of the wall of the uterine artery branches, after surgical procedures such as cesarean, birth dilatation and curettage, hysterectomy, or myomectomy [ [4] , [5] , [6] , [7] , [8] , [9] , [10] ]. Since UAP presents with per vaginal bleeding, timely diagnosis and management is important to prevent further blood loss and stabilize the patient's hemodynamics. Establishing a diagnosis can be difficult, radiological tests like ultrasonography (USG) and computed tomography (CT) angiography are required to visualize the aneurysm, which is not always conveniently accessible. Hence, UAP may go unnoticed as a possible cause of PPH. We report a case of UAP in a 25-year-old female which was successfully managed with embolization, to emphasize the importance of considering UAP as a potential etiology of PPH. Our work has been reported as per the SCARE Guidelines 2023 criteria [ 11 ].

Coi Statement

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

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