Postpartum spontaneous rupture of an ovarian artery aneurysm: A case report and review

Hui-Fen Yin; Hai-Yan Liu; Wen-Tao Jin; Rong Hu

doi:10.4103/2096-2924.296545

CASE REPORT

Year : 2020 | Volume : 4 | Issue : 3 | Page : 191-193

Postpartum spontaneous rupture of an ovarian artery aneurysm: A case report and review

Hui-Fen Yin¹, Hai-Yan Liu¹, Wen-Tao Jin², Rong Hu¹
¹ Department of Obstetrics, Obstetrics and Gynecology Hospital, Fudan University, Shanghai 200011, China
² Department of Radiology, Obstetrics and Gynecology Hospital, Fudan University, Shanghai 200011, China

Date of Submission	11-May-2020
Date of Decision	10-Jun-2020
Date of Acceptance	23-Jun-2020
Date of Web Publication	29-Sep-2020

Correspondence Address:
Rong Hu
Department of Obstetrics, Obstetrics and Gynecology Hospital, Fudan University, No. 419 Fangxie Road, Shanghai 200011
China

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/2096-2924.296545

Abstract

This study reported a case of spontaneous rupture of an ovarian artery aneurysm 4 days following cesarean section. The patient mainly presented with a consistent low-grade fever and did not have symptoms, such as severe abdominal or flank pain, as is common in other similar cases. Enhanced magnetic resonance imaging revealed a massive retroperitoneal hematoma; angiography confirmed that there was a rupture in an ovarian artery aneurysm. A selective embolization was performed successfully.

Keywords: Embolization; Ovarian Artery Aneurysm; Pregnancy; Retroperitoneal Hematoma

How to cite this article:
Yin HF, Liu HY, Jin WT, Hu R. Postpartum spontaneous rupture of an ovarian artery aneurysm: A case report and review. Reprod Dev Med 2020;4:191-3

How to cite this URL:
Yin HF, Liu HY, Jin WT, Hu R. Postpartum spontaneous rupture of an ovarian artery aneurysm: A case report and review. Reprod Dev Med [serial online] 2020 [cited 2020 Oct 28];4:191-3. Available from: https://www.repdevmed.org/text.asp?2020/4/3/191/296545

Introduction

The spontaneous rupture of an ovarian artery aneurysm is an extremely rare occurrence. As pregnancy-related ruptures are more complex and rare, they can be easily misdiagnosed; only 12 cases have been reported in the English literature during the past 30 years. Herein, we report a case of rupture of an ovarian artery aneurysm in the early postpartum period after cesarean section.

Case Report

A 38-year-old woman, gravida 6, para 2, was admitted to the hospital with a history of two cesarean surgeries and one right salpingectomy. The patient was pregnant with twins and delivered via cesarean surgery at 37 weeks of gestation and 1 day. All physical examinations were normal. The surgery went well, nothing special was found intraoperatively, and her vital signs were stable after the surgery. The patient's hemoglobin level before surgery was 103 g/L, and the 1^st day after surgery, a routine blood test showed that her hemoglobin level was 116 g/L, white blood cell count was 19.04 × 10⁹/L, and neutrophil was 82%.

However, 4 days after surgery, the patient was still experiencing a low-grade fever with a high temperature of 37.9°C. Another blood test showed that her hemoglobin level was 88 g/L, white blood cell count was 12.48 × 10⁹/L, and neutrophil was 71%. A pelvic ultrasound was performed and suggested that there were no unexpected postpartum findings. Since the white blood cell count was still high, the patient's antibiotics were changed to piperacillin sodium and tazobactam sodium for reducing the infection. Three days later, the fever had not improved, and the patient complained of right flank soreness. An abdominal examination showed right lower quadrant tenderness without rebound.

A blood test and pelvic ultrasound were performed again. The results showed that the hemoglobin level had continuously dropped to 75 g/L and that there may be a massive hematoma measuring 14.8 cm × 7.6 cm × 12.1 cm. Enhanced magnetic resonance imaging (MRI) of the abdomen and pelvis confirmed a huge mass measuring 19.1 cm × 13.5 cm × 8.0 cm at the right side of the uterus [Figure 1]. Multiple tortuous and dilated blood vessels were seen inside the mass with an enlarged artery having a 1.8-cm diameter.

Figure 1: Enhanced magnetic resonance imaging of the abdomen and pelvis. The transverse plane showing a large hematoma (transverse arrow) on the right side of the uterus with tortuous vascular structures inside (vertical arrow).

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An emergent angiography was performed to locate the origin of bleeding by catheterization through a right femoral approach with 5F catheters. It showed a tortuous right ovarian artery with segmental dilatations, confirming the presence of an aneurysm in the ovary [Figure 2]. A selective embolization of the right ovarian artery using gelatin sponge particles was performed. After embolization, we upgraded the patient's antibiotics to meropenem and monitored the patient's temperature, hemoglobin level, and size of hematoma. It showed that the patient's hemoglobin level stopped dropping, and her temperature stabilized after 5 days. The patient was discharged 11 days after embolization, and her hemoglobin level was back up to 101 g/L. Three weeks after embolization, an ultrasound showed a shrinking hematoma measuring 5.5 cm × 4.1 cm × 3.7 cm. At present, the patient is nearly 2 months postpartum and has not yet begun menstruation because of lactating. We plan to follow her laboratory analysis and ultrasound.

Figure 2: Right ovarian artery aneurysm (arrow) confirmed by angiography.

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Discussion

Pregnancy-related rupture of arterial aneurysms occurs in the decreasing order of the intracranial arteries, aorta, splenic artery, renal artery, and ovarian artery.^[1] Rupture of an ovarian artery aneurysm is extremely rare with currently only 12 reports in the English language literature in the past three decades, which are listed below in [Table 1].^{[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13]} From the literature review, it is clearly that all patients were in their thirties, multigravida, and mostly with unremarkable medical and obstetrical histories. In seven of the 13 cases (including ours), the aneurysm was on the right side, and 10 of the aneurysm ruptures occurred in the early postpartum period, two occurred in the third trimester of pregnancy, and one occurred during delivery. All of the aneurysm ruptures occurred in the postpartum period were after vaginal delivery. It was only in our case that the rupture occurred 4 days after a cesarean section.

Table 1: Reported pregnancy.related ovarian artery aneurysm rupture

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The mechanisms of the formation and rupture of ovarian aneurysm related to pregnancy are still not fully understood but are thought to be related to hemodynamic and hormonal changes during pregnancy. Physiological changes during pregnancy, including increased utero-ovarian perfusion, and the pressure of the gravid uterus applied to the aorta may induce structural changes in the arteries and may contribute to the formation of an aneurysm. In addition, segments of the ovarian arteries may fail to involute after pregnancy, thus predisposing aneurysm formation in a subsequent pregnancy.^[5] These cumulative changes made multiparous women a risk factor for the development of an ovarian artery aneurysm. Hormonal changes can also lead to weakening of connective tissue due to depolymerization of the collagen and loss of elastin, thereby promoting the rupture of an aneurysm. The relation between the rupture of ovary aneurysm and the route of delivery was rarely discussed. It is unclear that whether the stress of labor played a part in the rupture of aneurysm since some reviewers suggested that bleeds may occur initially during delivery and are only recognized if the bleed progresses and the patient becomes symptomatic.^[13] However, our case showed that the aneurysm rupture could also occur in the postpartum period after a cesarean section, the main mechanism may still due to the hemodynamic and hormonal changes, including failure of puerperal involution of a segment of the ovary artery. The underlying mechanism still need to be further explored.

The most common presenting symptom of a rupture is acute abdominal or flank pain with no prodromal symptoms. If symptoms appear in the third trimester, placental abruption and uterine rupture may be falsely diagnosed. However, in our case, the patient mainly presented with a consistent low-grade fever without obvious abdominal or flank pain. During the postpartum period, especially after cesarean section, these symptoms may be confused with surgical fever or uterine contraction pain, posing diagnostic difficulties. Due to consistent low-grade fever and drop in hemoglobin levels, a high index of suspicion for hematoma-related fever must be employed, and imageological examination should be performed actively. The diagnosis should be kept in mind, especially for multiparous women.

The treatment principle of ovarian aneurysms is to locate and manage the bleeding. Although a hematoma can be confirmed by computed tomography or MRI, it may be difficult to locate the hemorrhage precisely. Angiography can precisely determine the location and treat vascular abnormalities simultaneously, allowing us to avoid invasive laparotomy. Thus, transcatheter arterial embolization (TAE) has now become a useful alternative procedure in recent years. However, it could only be used when the patient is hemodynamically stable and requires experienced interventional radiologists. In our case, the patient is hemodynamically stable and interventional radiologists were available; thus, TAE was selected. In addition, TAE cannot remove collected blood and a remaining hematoma may carry a risk of infection; thus, clinicians should be aware of fever after TAE.

In conclusion, spontaneous rupture of an ovarian artery aneurysm is extremely rare and is often associated with nonspecific clinical signs but can be life-threatening and should be taken seriously. Pregnancy-related ruptures are more complex and can be misdiagnosed. This atypical case suggests that attention should be given to patients with consistent low-grade fever even without presentation of abdominal pain, and both imageological and blood examinations should be performed actively. Awareness of this issue may be help to diagnose and treat this problem earlier.

Acknowledgment

We wish to thank the ultrasound and radiologist team of professionals who were involved in the successful outcome of this case.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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