Case Report

Rupture of splenic artery aneurysm during early pregnancy: a rare and catastrophic event

Abstract

Spontaneous rupture of splenic artery aneurysm during pregnancy is a rare, life-threatening event with a catastrophic prognosis. Splenic artery aneurysm, known to be more frequent in women, especially among multiparous ones, is generally asymptomatic until rupture. Because of increased blood flow and hormonal modifications, this rupture occurs frequently during pregnancy, most often at the end of the third trimester. We present the case of a second parous woman in early pregnancy with a rupture of splenic artery aneurysm initially diagnosed as a complicated Ectopic pregnancy, which profoundly modifies surgical treatment.

Through this case report, we want to draw attention to the fact that even if complicated ectopic pregnancy is much more frequent, ruptured splenic artery aneurysm (SAA) needs to be considered as a part of differential diagnosis of hemoperitoneum during the first trimester as well.

Splenic artery aneurysm in pregnancy is a rare clinical entity that carries the risk of rupture, with fatal consequences for both mother and fetus. It is more frequent during the third trimester of pregnancy, as most of the cases have been reported during this period.

The clinical picture has been named “splenic emergency syndrome.” This clinical entity is due to a brisk intraperitoneal bleeding, leading to an abrupt onset of shock and a sudden unexpected death. Emergency physicians and other frontline staff must consider this potential condition in the differential diagnosis of severe upper abdominal pain in pregnancy.

The husband of a 35-year-old woman called the out-of- hospital Medical emergencies service after his wife experienced spontaneous onset of pain on the left side of the abdomen with vomiting, which had begun 3 hours earlier. Less than 10 minutes later, while the medical team was on its way, he called back because she became unconscious. The medical team arrived 15 minutes after the first call and found the patient in a severe state of hypovolemic shock with a blood pressure of 60/40 mm Hg, a heart rate of 150 bpm, and Glasgow Coma Scale of 7. Clinical examination revealed a distended abdomen with

signs of peritoneal irritation; immediate ultrasound scanning showed an abundant leakage of liquid into the peritoneal cavity. While the first resuscitation maneuvers were initiated, questioning of her husband revealed that she had not experienced any abdominal trauma the days before. Moreover, she was at 6 weeks of amenorrhea, confirmed by a recent quick urine test. Therefore, complicated extra- uterine pregnancy was highly suspected. During transport to the hospital, she was resuscitated with 2000 mL of colloid through 2 peripheral intravenous catheters, continuous norepinephrine infusion, and orotracheal intubation. Despite a slight Hemodynamic improvement, she remained extre- mely unstable. The surgical team was warned of her arrival and blood was ready to be transfused.

Taken directly to the gynecologic operating theater, she underwent an exploratory laparotomy that confirmed the massive hemoperitoneum, but the latter did not have a gynecologic origin. The arterial bleeding was actually coming from the lesser sac. Intensive care procedures with massive blood transfusion were carried on while the vascular team was called for reinforcement. A ruptured SAA was found and treated by proximal ligation of the splenic artery and splenectomy. Two hours later, as she started developing a severe disseminated intravascular coagulation, she died of sudden cardiac arrest in the intensive care unit, for which no clear explanation could be given.

Even if SAA is the most frequent among visceral artery aneurysms (more than 50%), it remains rare and generally asymptomatic until rupture [1,2]. Its real prevalence is unknown because diagnosis is usually fortuitous at the occasion of an autopsy or angiography; indeed, less than 5% of the patients with SAA present symptoms [3]. Except in patients with cirrhotic portal hypertension, rupture of SAA occurs more frequently among multiparous women, espe- cially during the third trimester of pregnancy [4,5]. When rupture occurs, a high mortality of 25% is reported [6]; this rate increases hugely to 75% among pregnant women, with an even higher 95% of fatal outcome for the fetus [7]. The increased incidence of SAA during pregnancy can be explained by 2 different mechanisms. The first one is endocrine and involves estrogen, progesterone, and relaxin, whose rising levels may lead to alteration of the structure of the arterial wall, which leads to fibromuscular dysplasia [8,9]. The second one is mechanical; pregnancy is

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accompanied by physiological changes that significantly increase the likelihood of aneurysm formation and rupture: higher blood volume, increased cardiac output, and portal hypertension due to uterine compression.

Sixty-nine percent of ruptured SAAs during pregnancy were reported during the third trimester, followed by second trimester and during labor for 13% [10]. Rupture during the first trimester has been reported only once in the literature so far [11]. As rupture of an SAA remains an uncommon cause of hypovolemic shock during late pregnancy, making this diagnosis is even more difficult in early pregnancy because complicated extrauterine preg- nancy is a more likely cause of hemoperitoneum.

This case report aims to warn the physician that hemoperitoneum during the first trimester is not exclusively caused by ectopic pregnancy. Ruptured SAA should be kept in mind as a differential diagnosis despite its low frequency because its management involves a different surgeon. That is why women in early pregnancy presenting with life- threatening, nontraumatic hemoperitoneum should be oriented to centers where a general or vascular surgeon is also available.

Acknowledgment

This work is attributed to SAMU31, Toulouse, France.

Julien Chookun MD

Pole d’anesthesie reanimation

GRC BP 48

Universite Paul Sabatier Toulouse, France

E-mail address: [email protected]

Vincent Bounes MD

Pole d’anesthesie reanimation

GRC BP 48

Universite Paul Sabatier Toulouse, France

Jean Louis Ducasse MD

SAMU 31

Pole de medecine d’urgences Hopitaux Universitaires

31059 Toulouse cedex 9, France

Olivier Fourcade MD, PhD

Pole d’anesthesie reanimation

GRC BP 48

Universite Paul Sabatier Toulouse, France

doi:10.1016/j.ajem.2008.10.039

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