Sudden Rupture of Splenic Artery Aneurysm in a Pregnant Woman Resulting in the Death of the Fetus: A Case Report and Review of Literature

Key content Abdominal pain in pregnancy is common, with a differential diagnosis that can encompass obstetric, surgical and medical conditions. Presentation of common surgical problems can be atypical in pregnancy, potentially delaying diagnosis. Surgical causes of abdominal pain to consider in pregnancy include appendicitis, cholecystitis, bowel obstruction, ureteric obstruction, pancreatitis and aneurysm rupture, most commonly involving the splenic artery. The management of surgical conditions in pregnancy requires continuing evaluation and potential modification to balance the medical, surgical and obstetric challenges. Management of the pregnant woman with a surgical cause of abdominal pain requires collaborative, multispecialty practice to optimise care of the mother and baby. Learning objectives To highlight the differential diagnoses of abdominal pain in pregnancy. To develop a structured assessment process for pregnant women with abdominal pain. To update the obstetrician on the priorities of surgical management for abdominal pain in pregnancy. Ethical issues Evaluating the risks of surgical interventions on the mother and baby with the high potential for premature delivery.

Rupture of splenic artery aneurysm (SAA) is a rare condition that mostly occurs during pregnancy and carries a potentially high maternal and fetal mortality. A 24 year-old female in her 20th week of gestation presented with epigastric abdominal pain and hemorrhagic shock related to spontaneous rupture of a large SAA in the mid portion of the splenic artery. The aneurysm was successfully excluded from circulation with endovascular placement of a covered stent-graft. This technique is less invasive than open surgical approach and maintains the patency of splenic artery, which is preferred over endovascular embolization.

EDITORIAL COMMENT: We accepted this rather detailed case report for publication because of the special relationship of rupture of splenic artery aneurysms with pregnancy. It is wise for obstetricians to know that this condition is an important uncommon cause of abdominal pain and collapse during pregnancy, labour or the puerperium.Summary: Rupture of a splenic artery aneurysm during pregnancy or delivery is a rare event. Only 99 cases have been reported. There is significant risk to mother and fetus. This paper describes a splenic artery aneurysm rupture with a concomitant unruptured hepatic artery aneurysm and coincidental partial placental abruption with fetal death at 22 weeks' gestation. Splenectomy was performed to achieve haemostasis. Nonsurgical treatment by embolization of the hepatic artery aneurysm was performed after a routine postoperative computed tomogram (CT) and angiogram revealed the lesion. CT screening of high‐risk patients and ultrasound/pulsed Doppler screening of pregnant women with unexplained abdominal pain might be an important precaution in the avoidance of obstetric catastrophies.

INTRODUCTION: Splenic artery aneurysm (SAA) is the third most common intraabdominal aneurysm. Splenic artery aneurysm rupture is a rare, but life-threatening complication of pregnancy. We present a case of splenic artery aneurysm rupture in labor where prompt diagnosis with multidisciplinary intervention resulted in a favorable maternal and fetal outcome. METHODS: A 24-year-old woman, gravida 1 para 0 at 39 weeks of gestation presented to labor and delivery unit for a scheduled induction of labor for gestational hypertension. During the course of induction of labor, patient had an abrupt onset of nausea and chills associated with hypotension and tachycardia. Fetal heart rate deceleration was noted and the patient was taken for a stat cesarean section due to persistent fetal bradycardia and worsening maternal clinical status. RESULTS: Intraoperatively significant hemoperitoneum was noted with no evidence of uterine rupture. Further abdominal exploration by the surgical team revealed a splenic artery aneurysm rupture. Patient underwent a splenectomy and splenic artery ligation. The patient was stabilized and transferred to the intensive care unit for further monitoring. CONCLUSION: Splenic artery aneurysm rupture is associated with a very high maternal and fetal mortality of 75% and 95% respectively. The prevalence of splenic artery aneurysm is noted to be 0.1–0.2% in general population from autopsy studies. The changes associated with pregnancy increase the risk of formation and rupture of the aneurysm. Symptoms are nonspecific and the diagnosis is often times made during laparotomy. Prompt diagnosis and a concerted team effort is essential as in this case for maternal and fetal survival.

Although splenic artery aneurysm (SAA) rupture in the pregnant patient is considered a rare occurrence, its consequences can be catastrophic for both mother and fetus. The mortality rate from this entity in pregnancy is disproportionately high when compared with that of the general population, with multiparity thought to be a contributing factor. Potential reasons for this may be explained by its non‐specific abdominal symptoms often misdiagnosed as common obstetric differentials, combined with a rapid deterioration of patient condition post rupture. For these reasons, SAA rupture should have a high index of suspicion when a pregnant women presents with an acute abdomen, as a favourable outcome is dependent on prompt diagnosis and appropriate medical intervention. This article reviews the incidence of SAA rupture in pregnancy, outlines the pathophysiological processes that contribute to this entity being more prevalent in the gravid population, and displays the pivotal role ultrasound can play in expediting this diagnosis. In the emergency situation, ultrasound examination of a pregnant patient should be extended from a focussed pelvic study to encompass the entire abdominal cavity. Its portability combined with its nonionising nature makes ultrasound the modality of choice to reduce the time to diagnosis and thus influence treatment pathways.

Rupture d’un anévrisme de l’artère splénique en fin de grossesse : à propos d’un cas et revue de la littérature

IntroductionPregnancy has been demonstrated as a significant risk factor of splenic artery aneurysm (SAA) formation and rupture. However, prompt diagnosis of SAA rupture in a pregnant patient showing acute abdomen has been practically challenging in light of its rarity and vague initial presentation. Presentation of caseA 40-year-old woman (gravida 1, para 0) at 35 weeks’ gestation presented to the emergency department with upper abdominal pain and nausea. Because of fetal dysfunction, emergency caesarian section was performed by a Pfannenstiel incision. Following delivery, 400 g of hemorrhage was removed from the upper abdominal cavity. Computed tomography showed a 37-mm SAA associated with copious adjacent fluid. Although selective angiography did not demonstrate active extravasation, interventional isolation of the SAA was not performed because of multiple surrounding arteries. Relaparotomy with an upper midline incision was then performed. Sudden cardiac arrest occurred upon opening the lesser sac to irrigate clots, and cardiac massage and proximal and distal clamping of the SAA were required. Eventually, splenectomy with excision of the SAA and pancreatic tail was successfully performed, but gauze packing of the open surgical wound was required because of severe coagulopathy. Following removal of the packs and closure of the abdomen 2 days after splenectomy, the patient and infant satisfactorily recovered without sequelae. DiscussionGiven continual awareness of abdominal vascular collapse during pregnancy, undelayed diagnosis and safer intervention might be achieved. ConclusionAwareness at initial presentation and multidisciplinary efforts might be essential to achieve maternal and fetal survival in SAA rupture during pregnancy.

Sir, We wish to call attention to the case of a 39-year-old woman, pluripara, in her 35 weeks’ gestation who presented acute abdominal pain with vomiting. The past clinical and obstetric history was unremarkable. The pain was dull and gradually increasing, mainly located to the epigastric and left upper quadrant. Blood pressure and heart rate were normal. Ultrasound examination was performed showing normal fetal heart rate and biometry and a considerable collection of free-fluid and blood clot in the pouch of Douglas. The patient developed severe hypotension, acute anemia and syncope. An emergency cesarean section was performed and a massive hemoperitoneum confirmed. The uterus was intact and there was no visible bleeding from any other pelvic organ. A live fetus (Apgar score 4–8) was delivered and was transferred to the Neonatal Intensive Care Center. The placenta did not show evidence of abruption. Exploration of the abdomen with a general surgeon in attendance showed a ruptured splenic artery aneurysm located in the distal segment of the vessel. The aneurysmal sac was conservatively treated by surgical ligature of the splenic artery. Bleeding was controlled with prompt recovery of the blood pressure. Careful inspection of the spleen was undertaken and no signs of ischemic areas were noted and it was decided to avoid splenectomy meantime. A postoperative total body computed tomography scan was done to confirm possible residual bleeding and/or ischemic areas in the spleen, but both were negative. The patient received three units of red blood cells and was discharged home with her baby 10 days after surgery. Splenic artery aneurysm is the most common visceral artery aneurysm (1–3). It occurs more frequently in younger females. Up to 95% of cases take place during pregnancy, particularly in third-trimester multiparous women, considered to be related to endocrine and mechanical causes (3). Most are asymptomatic until rupture. A recent review reports 32 cases showing a mean maternal and gestational age of 27.9 years and 34.5 weeks, respectively (3). In our case the distal part of the splenic artery was involved, which is rare (3). Splenic artery rupture in pregnancy is rare and can be catastrophic, with high maternal and fetal mortality (1–3). Splenectomy is usually performed in addition to resection and/or ligation of the aneurysm (2) but may sometimes be avoided because of multiple peripancreatic and perisplenic collateral vessels that enable perfusion even after splenic artery ligation. The development of such a vascular variant was demonstrated by magnetic resonance imaging six months after surgery. Obstetricians should consider possible rupture of a splenic artery aneurysm as a cause of acute abdominal hemoperitoneum in the third trimester of pregnancy.

Loss of renal India ink artifact—a useful radiological sign for obstructive hydronephrosis in pregnancy

Introduction: Splenic artery aneurysms (SAAs) are the most common (60%) of visceral artery aneurysms with the possibility of rupture. SAA rupture is associated with 20% mortality, which increases disproportionately to 75% in pregnant women, with fetal the mortality rate of 95%. Aim: We present a discussion based on the case report with a review of the literature on ruptured SAA in pregnancy as well as possible treatment options, complications and prognosis. Case study: Patient, 40-year-old pregnant woman, gravida 2, para 1, 33 week of pregnancy, with ruptured SAA, underwent cesarean section, splenectomy, and hypovolemic shock treatment. The outcomes of the mother and neonates were good. Postoperative recovery was complicated by right-sided pneumothorax. Results and discussion: 87% of patients with SAA are females. In our analysis of cases, with ruptured SAA in pregnancy, the parity may be the factor that influences the maternal and fetal outcomes. In third trimester of pregnancy (as in our case study) the most often mistaken indication for laparotomy was a suspected placental abruption. Conclusions: Ruptured SAA during pregnancy should be considered as one of the non-obstetric causes of intraabdominal bleeding. Careful observation of the patient from the onset of symptoms is a key approach that increases maternal and fetal survival.

Dear Editor, Thrombotic thrombocytopenic purpura (TTP) is a rare complication of pregnancy with a poor prognosis and a high fetal mortality when presenting early during gestation. Few cases of TTP (10%) occur during the first trimester [1, 2], the majority occurring at the time of delivery or during post-partum period [4, 6]. Until the effectiveness of plasma infusion and plasma exchange was recognized, TTP was associated with a mortality rate of 95% and, in case of pregnancy-related TTP, the maternal survival was rare and the fetal mortality rate approached 80% [9]. Fetal death is secondary to placental infarction due to thrombotic occlusion of the decidual arterioles [8]. Currently, plasma infusion and plasmapheresis have improved maternal and fetal survival rates [10]. At present, it is well known that termination of pregnancy is rarely needed and that the fetal survival rate could be improved by effective maternal therapy [14]. When TTP occurs during the first trimester of pregnancy and delivery of a viable infant is not an option, plasma exchange treatment is urgently indicated and may allow the pregnancy to continue [13]. There are some reports showing that prolonged courses of plasma exchange, beginning as early as 6 weeks of gestation, may achieve and maintain a remission of TTP allowing delivery of a healthy, full-term infant [1]. When plasma exchange fails to induce remission, therapeutic abortion may be considered, although the response of TTP to termination of pregnancy is uncertain. We have previously reported a case of a successful pregnancy in a young female who developed a severe TTP relapse at 18 weeks of gestation [5]. In the present study, we describe a case of a first pregnancy concomitant to a severe TTP diagnosed in the first trimester with maternal survival and delivery of a healthy child. In August 2004, a 26-year-old nulliparous woman at 9 weeks of gestation presenting macrohematuria and ecchymoses was admitted to our institution. Laboratory data showed thrombotic microangiopathy, with anemia (7.5 g/dl), thrombocytopenia (platelets 6×10/l), numerous schistocytes (60%) in the peripheral blood smears, elevated levels of lactic dehydrogenase (1,472 IU/l range 70–190) and bilirubin (total 3.4 mg/dl, normal value 0.4–1.1, indirect 3 mg/dl, normal value 0.1–0.4), reticulocytosis (250×10/l), and negative direct/indirect Coombs’ test. Renal function was normal. Bone marrow aspirate showed a normal bone marrow cytomorphology. Thrombophilic screening showed normal levels of antithrombin (AT), absence of lupus anticoagulant (LAC), normal levels of C and S proteins, and no mutations of Leiden Factor V and prothrombin gene, while a heterozygous mutation of MTHFR C677T, with normal plasma homocysteine concentrations, was found. Serologic examinations were negative for anti-extractable nuclear antibodies and anticardiolipin antibodies. Unfortunately, ADAMTS-13 levels and inhibitor were not determined during the acute event. On the basis of the symptoms and laboratory tests, a clinical diagnosis of TTP was made and the patient was treated with intravenous methylprednisolone (2 mg/kg/die to 120 mg/die) and daily plasmapheresis, with plasma replacement of 30 ml/kg/die, in combination with continuous infusion of fresh frozen plasma (FFP) (10 ml/kg/die). Folic acid (15 mg/die) support was given. The initial response was Ann Hematol (2009) 88:287–289 DOI 10.1007/s00277-008-0579-4

Adrenal infarction can be hemorrhagic or non-hemorrhagic, while the latter is much less common. Non-hemorrhagic adrenal infarction (NHAI) is a rare but potentially serious cause of acute abdominal pain in pregnancy that can lead to significant adverse outcomes for both mother and baby if not treated promptly and appropriately. Diagnosis of this condition is challenging due to the non-specific nature of clinical and laboratory findings. Ultrasound (US) has limited utility in diagnosing retroperitoneal pathology, while the use of computed tomography (CT) is restricted due to concerns about fetal radiation exposure. Diagnosis is typically based on magnetic resonance imaging (MRI) findings, with other potential etiological causes of abdominal pain being systematically ruled out. Treatment primarily consists of analgesics and anticoagulants. In this case report, we present a pregnant patient who arrived at the emergency department with an acute abdomen. Various MR images are provided, illustrating the diagnostic process. The patient was successfully treated with anticoagulants and antibiotherapy. Through this case, we aim to highlight the role of imaging in the differential diagnosis, management, and follow-up of pregnant patients presenting with acute abdominal conditions.

Rectus sheath hematoma (RSH) is a rare, but important potential cause of acute upper abdominal pain in pregnancy. We report a patient with history of hemolysis, elevated liver enzymes, low platelets syndrome who presented with upper abdominal pain, hypotension, elevated hepatic transaminase levels, and a visibly enlarging right costal margin mass at 33 weeks' gestation. She underwent emergent cesarean delivery and upper abdominal exploration with findings of a large hematoma involving the entire right rectus sheath. The current case illustrates that the diagnosis of an RSH in pregnancy can be difficult as its presentation can mimic many common causes of abdominal pain seen outside of pregnancy, as well as many specific to pregnancy. Review of the published cases from the past 20 years shows that correct diagnosis remains difficult in pregnancy despite advances in diagnostic imaging. Although management of an RSH outside of pregnancy is frequently conservative, it is very often treated surgically in pregnant patients and often leads to preterm delivery.

Splenic artery aneurysm (SAA) is the most common (60%) of all visceral artery aneurysms. The majority of these cases are asymptomatic, but the presentation of their rupture can vary from abdominal/chest pain to cardiovascular collapse (Sadat U, Dar O, Walsh S, Varty K. Splenic artery aneurysms in pregnancy-a systematic review. Int J Surg. 2008;6(3):261-265.). Although rare, the mortality associated with the rupture is as high as 25% (De Vries JE, Schattenkerk ME, Malt RA. Complications of splenic artery aneurysm other than intraperitoneal rupture. Surgery. 1982;91(2):200-204; Caillouette JC, Merchant EB: Ruptured splenic artery aneurysm in pregnancy. Twelfth reported case with maternal and fetal survival. Am J Obstet Gynecol 1993;168(6 Pt 1):1810-1811) and increases to 75% among pregnant women with a concomitant fetal mortality of 95% (O'Grady JP, Day EJ, Toole AL, et al. Splenic artery aneurysm rupture in pregnancy. A review and case report. Obstet Gynecol. 1977; 50(5):627-630). Because of such high maternal and fetal mortality prompt management of SAAs is of utmost importance. We are presenting a case of a 35-year-old woman with a missed ruptured SAA who after an emergent cesarean section went into profound shock and was unable to be resuscitated. This case illustrates the importance of considering the diagnosis of SAA rupture in hemodynamically unstable peripartum females.

The spleen