A Case of Spontaneous Ovarian Artery Haemorrhage in a Post-Menopausal Woman with Congenital Solitary Kidney

By Lu Min Chen, Feng Jiang, Hongjie Hu

Affiliations

1. Department of Radiology, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, Hangzhou, Zhejiang, China

doi: 10.29271/jcpsp.2022.Supp.S137

ABSTRACT
Ovarian arterial haemorrhage (OAH), a rare cause of spontaneous retroperitoneal hematoma (SRH), usually occurs in women during pregnancy or in those with a history of repeated pregnancies. The clinical manifestations of OAH are non-specific; hence, proper and timely imaging examinations are extremely important. Contrast Enhanced CT scan is the first choice for clarifying the cause of haemorrhage in patients with SRH. Trans-arterial embolisation (TAE) has become the most common treatment option for OAH. Here, we report a case of SRH in the left ovarian artery region in a patient with a congenital solitary kidney. The patient was initially misdiagnosed as left kidney haemorrhage which was successfully embolised with tissue glue. There are no previous reports of OAH in patients with a congenital solitary kidney.

Key Words: Ovarian artery, Spontaneous retroperitoneal hematoma, Congenital solitary kidney, Trans-arterial embolization.

INTRODUCTION

Ovarian arterial haemorrhage (OAH) can cause spontaneous retroperitoneal hematoma (SRH) in rare cases, which may lead to serious clinical consequences. It should be considered in women with a history of repeated pregnancies and SRH during pregnancy. OAH tends to present with abdominal or low back pain, and the clinical manifestations are non-specific. It is possible to diagnose OAH with computed tomography (CT) scan, prior to digital subtraction angiography (DSA) or surgery. Trans-arterial embolisation (TAE) has become the most common treatment option for OAH.

We report a case of SRH in the left ovarian artery (OA) region in a patient with a congenital solitary kidney. Initially, the patient was misdiagnosed as left kidney haemorrhage and successfully embolised with tissue glue. OAH has never been reported in congenital solitary kidney patients.

CASE REPORT

A 50-year female came to our emergency room with an apparent left renal haemorrhage that started three days prior to admission. Before coming to our hospital, the patient had been treated conservatively for three days with no improvement.

Figure 1: (a) A plain CT scan of the abdomen suggests a large hematoma (white arrow) in the left retroperitoneum, occupying the left renal region and easily misdiagnosed as a ruptured haemorrhage of the left kidney; (b) A contrast-enhanced CT scan of the abdomen with coronal reconstruction shows a large hematoma in the left renal region extending down to the entrance of the pelvis (white arrow); (c) Abdominal enhanced CT suggests an aneurysm with enhanced hematoma margins (white arrow); (d) Abdominal enhanced CT: there was no image for the left kidney compared with the right kidney, and the opening of the left ovarian artery can be seen (white arrow). (e) Maximum intensity projection (MIP) in abdominal enhanced CT scan showed the left ovarian artery (black arrow) and aneurysm (white arrow).

There was no obvious cause for her symptoms at the time of onset. On physical examination, heart rate was 77 beats/min, blood pressure was 110/69 mmHg, and haemoglobin was 56 g/L. The pain was present in the left lumbar area, especially after renal percussion. Scattered longitudinal stretch marks could be seen in the lower abdomen. The patient had previously been in good health, with a history of two births, no miscarriages, and one year of menopause. There was no history of hypertension, diabetes mellitus, vasculitis, or other diseases that may have caused arterial damage. There was no recent trauma and the patient had never been administered anticoagulant medications. An emergency contrast-enhanced CT scan of the abdomen was performed, which revealed a massive hematoma (up to 14 cm in diameter) in the left posterior peritoneum occupying the left kidney region, with no signs of the left renal arteries and veins. Furthermore, normal enhanced renal parenchyma was not appreciated. Thus, we were convinced that the patient lacked the left kidney and discarded the original renal haemorrhage diagnosis. The edge of the hematoma was visible with an irregular shaped aneurysm which was 5 mm in diameter. After careful analysis of the bleeding artery source, the hematoma was considered to be due to OAH (Figure 1). An emergency DSA was performed.

Figure 2: (a) Abdominal aorta aortography shows the left ovarian artery opening (arrows); (b) Ovarian artery aneurysm (arrows); (c, d) Selective imaging of bilateral ovarian arteries; (e) A repeated abdominal aortography after left ovarian artery embolisation shows complete occlusion of the left ovarian artery, including the proximal and distal end of the aneurysm neck (arrows); (f) Coronal maximum intensity projection (MIP) reconstruction suggests that the hematoma was significantly absorbed and reduced (long black arrows) and retention of embolic material in the left ovarian artery (white arrows). The left ovarian artery was well-formed with the lateral branches of the left uterine artery and that there was no ischaemia in the left ovary.

After the right femoral artery was successfully punctured with Seldinger's technique, a 5 F sheath (Terumo®, Japan) was inserted. The left OA was selected with a 5 F Cobra catheter (Cordis®, America) and superselected with a 2.7 F microcatheter (Progreat®; Terumo®, Japan). The left OA aneurysm was detected by angiography and was successfully embolised with tissue glue (Histoacryl®; Braun, German) diluted with iodine oil (Lipiodol®; Guerbet, France) (Figure 2). The ratio of Lipiodol to tissue glue was 9:1. The rest of the treatment included aggressive hydration, iron supplementation, and administration of antibiotics. The patient was discharged 5 days after TAE with no procedure-related complications. Two months after the surgery, a follow-up CT scan showed significant absorption of the left SRH, retention of glue in the left OA, and patency of the distal OA, suggesting no ischaemia in the left ovary (Figure 2).

DISCUSSION

There is considerable variation in the anatomy of the OA, which can originate from the horizontal abdominal aorta at the L1-L4 level. The OA usually opens anterolateral to the abdominal aorta at the level of L2, between the renal and sub-mesenteric artery openings, and to a lesser extent in the renal artery; its trunk travels outward in a spiral along with the psoas major muscle down to the pelvis, then traverses inward to the ovarian portal to supply the ovaries and anastomoses with the uterine artery.¹

During pregnancy, aneurysms often occur in the aorta, intracranial artery, splenic artery, renal artery, coronary artery, and OA.² OA aneurysms are sporadic and usually asymptomatic. Thus, they are commonly found during clinical examinations. Once rupture occurs, it can lead to severe retroperitoneal bleeding, which can be life-threatening.

The pathogenesis of OAH is currently poorly understood. According to the literature, it is believed to be related to repeated pregnancies. ³ Since OAH often occurs during pregnancy or the postpartum period, it is speculated that it may be related to hemodynamic and endocrine changes during pregnancy. Repeated pregnancies, accompanied by fluctuating blood pressure, mechanical pulling during delivery and surgery, and subsequent rupture and bleeding,³ set the conditions for the development and progression of the aneurysms. There are often abundant anastomoses between the OA and the uterine artery, and many uterine fibroids can be supplied with blood by the OA. One report exists of an OAH caused by the OA providing the uterine fibroids.⁴ In addition, chronic vascular damage caused by hypertension and diabetes mellitus, as well as vascular inflammatory changes due to human immunodeficiency virus (HIV) have also been reported to be related to OAH.^5-7 In the present case, the left kidney was absent. Generally, a congenital solitary kidney does not affect the anatomy of the OA; therefore, we believe that there was no direct relationship between the OAH and the solitary kidney in this patient. In this case, OAH might be mainly related to the previous two pregnancies.

For patients with SRH, appropriate and timely imaging examinations are extremely important, since the clinical symptoms of OPH are non-specific and often present only as abdominal or lower back pain. In the present case, the patient was originally misdiagnosed as left kidney haemorrhage in another hospital, based solely on a plain abdominal CT scan. However, contrast-enhanced CT in our hospital found that the patient's left kidney was absent, suggesting the critical diagnostic value of enhanced CT. Enhanced CT has become the first choice for SRH diagnosis.^5,8 Although a CT scan is usually contraindicated for pregnant patients, enhanced CT should still be recommended in pregnant patients with unexplained SRH. When the cause of bleeding is difficult to determine with CT, further strategies such as angiography or surgical exploration should be considered. Colour doppler ultrasound may also be contemplated as an alternative when enhanced CT is not available, for it has been reported to be useful in diagnosing OAH.⁹

TAE is the current preferred method for managing hemodynamically stable OAH. Failure of OA super selection is rare and the few reports that exist are mostly due to tortuous or small openings of the OA.⁵ In order to avoid prolonged super selection, such patients should be promptly referred to surgery. In the past, laparotomy and vascular ligation were considered to be the first choice for the treatment of OA bleeding. Improvements in interventional therapy have made it the first treatment option for most hemodynamically stable patients. However, patients with difficult OA super selection, unstable hemodynamics, or the need for concurrent hematoma removal should be treated surgically.¹⁰

In the present case, we chose a 10% concentration of glue as the embolisation material because of three considerations: 1) Proper concentration of glue can easily cover the proximal and distal aspects of the aneurysm neck to reduce the backflow of blood; 2) From our own experience, 10% of glue cannot reach artery endings, which could cause ovarian ischaemia. In this case, the distal left OA was found to be unobstructed on re-examination. This finding suggested that the anastomotic branch between the distal left OA and the uterine artery was well-formed. Further, there was no ischaemia in the left ovary; and 3) tissue glue embolisation does not depend on the patient’s coagulation function and has been reported to be better than spring coils and gelatine sponges in patients with severe blood loss.¹¹

In conclusion, spontaneous haemorrhage from OA is extremely rare but may result in severe clinical consequences. A history of recurrent pregnancies or the presence of unexplained SRH in pregnant patients should alert to the possibility of OAH. An enhanced CT scan is the first choice for such patients. When available, emergency OA embolisation can be performed to stop bleeding.

PATIENTS^’ CONSENT:
Informed consent was obtained from the patient.

COMPETING INTEREST:
The authors declared no conflict of interest

AUTHORS’ CONTRIBUTION:
LMC: Data collection and write-up.
FJ: Literature review.
HH: Critical review and proofreading.
All the authors have approved the final version of the manuscript to be published.

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