CASE REPORT

Splenic Artery Aneurysm in Pregnancy Jacqueline Parrish,1 Cynthia Maxwell, MD, FRCSC, RDMS,2 John Robert Beecroft, MD, FRCPC3 Department of Biology, University of Toronto, Toronto ON

1

Department of Obstetrics and Gynecology, Maternal Fetal Medicine Division, Mount Sinai Hospital, University of Toronto, Toronto ON

2

Division of Vascular and Interventional Radiology, Department of Medical Imaging, Toronto General Hospital and Mount Sinai Hospital, University of Toronto, Toronto ON

3

Abstract Background: Splenic artery aneurysm (SAA), a rare condition chiefly affecting women, poses significant challenges for management when it occurs during pregnancy. Reports of successful management of SAA before rupture in pregnancy are limited, with several post-rupture cases reported. Case: We describe the case of a woman with an SAA of 13 × 9 mm near the hilum of the spleen who subsequently became pregnant. Embolization of the splenic artery in the third trimester resulted in occlusion of the aneurysm but was followed three weeks later by a splenic abscess. The patient went on to deliver a healthy baby. Conclusion: This case illustrates the importance of maintaining awareness of SAA in pregnancy because the condition carries a high risk of rupture and hemorrhage with high rates of fetal and maternal mortality.

Résumé Contexte : L’anévrisme de l’artère splénique (AAS) est une maladie rare qui affecte principalement les femmes et dont la prise en charge pose des défis considérables lorsqu’elle se manifeste pendant la grossesse. Les signalements de prise en charge réussies de l’AAS avant la rupture pendant la grossesse sont limités, plusieurs cas post-ruptures ayant été signalés. Cas : Nous décrivons le cas d’une femme présentant un AAS de 13 mm sur 9 mm près du hile de la rate qui est subséquemment devenue enceinte. Bien que l’embolisation de l’artère splénique au cours du troisième trimestre ait mené à l’occlusion de l’anévrisme, elle a été suivie par la manifestation d’un abcès splénique trois semaines plus tard. La patiente en est venue à accoucher d’un enfant en santé. Conclusion : Ce cas illustre l’importance de garder la présence d’un AAS à l’esprit pendant la grossesse, car cette pathologie donne lieu à un risque élevé de rupture et d’hémorragie qui se traduit en des taux élevés de mortalité fœtale et maternelle. J Obstet Gynaecol Can 2015;37(9):816–818 Key Words: Splenic artery, aneurysm, pregnancy Competing Interests: None declared. Received on December 15, 2014 Accepted on March 11, 2015

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INTRODUCTION

S

plenic artery aneurysm (SAA) is a rare condition occurring predominantly in women.1 Because the condition is commonly asymptomatic, identification of SAA is often incidental, at the time of ultrasound examination or autopsy.2 In pregnancy, the condition carries with it a high risk of rupture and hemorrhage, with a high rate of fetal and maternal mortality post-rupture.3 Surgical intervention is recommended in the first or second trimester because rupture most commonly occurs in the third trimester.4 Surgical intervention pre-rupture is rare, with most reports describing identification of the aneurysm post-rupture.5 The pathogenesis of the condition is not well understood, although an increase in circulating estrogen, progesterone, and relaxin levels during pregnancy is thought to increase blood flow and blood pressure within the splenic artery, weakening the arterial wall if there is pre-existing atherosclerosis and resulting in dilatation of the aneurysm.1,6 THE CASE

A 36-year-old Caucasian woman with two previous term pregnancies underwent an ultrasound examination for abdominal pain. She was suspected of having an SAA measuring 13 × 9 mm near the hilum of the spleen, and this was confirmed by subsequent CT scan (Figure 1). The patient was not pregnant at the time of diagnosis, but was considering a third pregnancy. She was counselled regarding the increased risk of rupture and the difficulty of management during pregnancy, and was also counselled regarding the options for management: splenic artery embolization, splenectomy, or expectant management. Nine weeks after the initial diagnosis, the patient presented at eight weeks’ gestation, and opted to undergo splenic artery embolization. A multidisciplinary team including specialists in maternal– fetal medicine and interventional radiology recommended

Splenic Artery Aneurysm in Pregnancy

Figure 1. 3-D reconstruction from CTA (obtained before pregnancy) demonstrating the 13 mm × 9 mm aneurysm of the splenic artery branch to the upper pole of the spleen, distal to the branch supplying the lower half of the spleen.

Figure 2. Fluoroscopic image obtained post coil embolization of the splenic artery aneurysm. Coils were placed proximal and distal to the aneurysm as well as within the aneurysm sac for occlusion. The arterial branch to the lower splenic pole was preserved.

deferring the procedure until 31 weeks’ gestation. The patient was also known to have gastroesophageal reflux disease and was regularly taking esomeprazole before pregnancy.

coils were also deposited within the aneurysm sac. A total of four 6 mm and two 10 mm Nester 0.018” platinum coils (Cook Medical, Bloomington, IN) were used. Following embolization, injection of contrast into the splenic artery demonstrated a good technical result, with occlusion of the upper pole branch of the splenic artery and non-filling of the aneurysm (Figure 2). Blood flow to the lower spleen was confirmed by demonstrating patency of the lower pole branch. The patient was subsequently discharged with no immediate post-procedural complication.

At 31 weeks’ gestation, the patient was admitted to hospital for the embolization procedure. Several measures were used to minimize radiation exposure to the fetus: most of the maternal abdomen was shielded with a lead apron, low-dose pulsed fluoroscopy was used, and no digital subtraction angiography or conventional radiographs were obtained. The patient was given corticosteroids to assist fetal pulmonary maturation in the event of preterm delivery, and intravenous cefazolin was administered prophylactically. The right common femoral artery was accessed in retrograde fashion and a 5 Fr vascular sheath was inserted. A 5 Fr SOS Omni catheter (Angiodynamics Inc., Queensbury, NY) was used to select the celiac artery. Coaxially, a 2.4 Fr Progreat microcatheter (Terumo Medical, Somerset NJ) was advanced along the tortuous splenic artery and into the branch to the upper pole of the spleen, distal to the aneurysm. Because of the wide neck of the aneurysm, selective embolization of the aneurysm alone (with preservation of flow in the splenic artery’s upper pole branch) was not possible. To occlude the aneurysm completely, the upper pole branch was embolized both proximal to and distal to the aneurysm, and embolization

Three weeks after the embolization, at 36+3 weeks’ gestation, the patient presented with chills, tachycardia, a temperature of 38.5°C, and irregular uterine contractions. Abdominal ultrasound imaging showed an 11-cm abscess in the upper pole of the spleen. The patient was admitted to hospital and a percutaneous drain was inserted. A volume of 300 cc of purulent, light brown, malodorous fluid was drained and sent for culture, with subsequent growth of coagulase negative Staphylococcus. The patient was given intravenous fluids, ampicillin, gentamicin, and metronidazole; however, the tachycardia persisted. The patient was transferred to the ICU with suspected early sepsis, and vancomycin was substituted for ampicillin. Her condition improved and stabilized over the SEPTEMBER JOGC SEPTEMBRE 2015 l 817

Case Report

next 48 hours, with normalization of her temperature and heart rate. Her uterine contractions had subsided. Blood and urine cultures carried out at the time of admission showed no growth. The patient was transferred from ICU to the labour floor, and after discussion the decision was made to proceed with induction of labour. This was successfully achieved with artificial rupture of membranes and use of intravenous oxytocin. She subsequently went on to have spontaneous vaginal delivery of a healthy female infant weighing 2720 g. Apgar scores were 9 at one minute and 9 at five minutes. Postnatal umbilical pH was 7.24 and arterial pH was 7.32. DISCUSSION

Most case reports concerning the management of SAA in pregnancy are post-rupture reports, with minimal guidance for patient management and intervention prerupture. During pregnancy, splenic artery aneurysms are prone to rupture in the third trimester or in the puerperium because of the heightened circulating levels of estrogen, progesterone, and relaxin, in addition to maternal physiological changes, which increase arterial stress.2 Previous reports describe high maternal (75%) and fetal (95%) mortality rates post-rupture, with only a small number of cases reporting survival for both the mother and the fetus.5 For pregnant women with an SAA ≥ 2 cm in diameter, minimally invasive surgical intervention is the recommended method of treatment.4 However, Lang et al. recommend that even an SAA ≤ 2 cm in diameter should be treated surgically in pregnancy, as the diameter of the aneurysm does not reflect the probability of rupture.3 Accepted minimally invasive surgical techniques for treating SAA consist of splenic artery ligation, arterial embolization, resection of the splenic artery, and splenectomy. Prophylactic administration of antibiotics is recommended for embolization involving the splenic artery.7 Post-rupture management is aimed at, and limited to, halting hemorrhage.6 While splenic artery aneurysms are generally asymptomatic, pregnant women who are experiencing pain in the left upper quadrant of the abdomen should be screened immediately for a possible SAA or other pathology.2 Often, the rupture of an SAA in pregnancy leads to misdiagnosis because it presents with symptoms (nausea, vomiting, hypotension) that are similar to more common obstetrical emergencies.3,6 When SAA is diagnosed during pregnancy, surgery is necessary to ensure an optimal outcome because of the high risk of rupture and high mortality rates.5

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We identified a systematic review4 and an institutional series8 describing outcome in rupture of SAA during pregnancy and postpartum, but could only identify a single article describing prophylactic treatment of a 2 cm SAA in a symptomatic woman.9 In this case, the aneurysm was diagnosed a year before pregnancy, but surgical resection was subsequently needed during the third trimester due to maternal symptoms including epigastric and left upper quadrant pain. Postoperative infections also pose a maternal and fetal risk and should be managed accordingly. CONCLUSION

Although SAA is rare in pregnancy, it is important to maintain awareness of this condition due to the high maternal and fetal mortality rate post rupture. It is also important to maintain awareness of this condition in pregnancy to avoid misdiagnosis. Immediate action is necessary following diagnosis to ensure optimal maternal and fetal outcomes. ACKNOWLEDGEMENT

The woman whose story is told in this case report has provided written consent for its publication. REFERENCES 1. Al-Habbal Y, Christophi C, Muralidharan V. Aneurysms of the splenic artery—a review. Surgeon 2010;8:223–31. 2. Lynch MJ, Woodford NWF. Rupture of a splenic artery aneurysm during pregnancy with maternal and foetal death: a case report. Med Sci Law 2008;48:342–5. 3. Lang W, Strobel D, Beinder E, Raab M. Surgery of a splenic artery aneurysm during pregnancy. Eur J Obstet Gynecol Reprod Biol 2002;102:215–6. 4. Sadat U, Dar O, Walsh S, Varty K. Splenic artery aneurysms in pregnancy—a systematic review. Int J Surg 2008;6(3):261–5. 5. Pavlis T, Seretis C, Gourgiotis S, Aravosita P, Mystakelli C, Aloizos S. Spontaneous rupture of splenic artery aneurysm during the first trimester of pregnancy: report of an extremely rare case and review of the literature. Case Rep Obstet Gynecol 2012:1–3. 6. Gourgiotis S, Alfaras P, Salemis NS. Spontaneous rupture of splenic artery aneurysm in pregnancy: a case report. Adv Med Sci 2008;53(2):341–3. 7. Moon E, Tam M, Nikano R, Karuppasamy K. Prophylactic antibiotic guidelines in modern interventional radiology practice. Semin Intervent Radiol 2010;27(4):327–37. 8. McMahon DP, Ward WH, Harwood JL, Moore EM. An institutional review of splenic artery aneurysm in childbearing-aged females and splenic artery aneurysm rupture during pregnancy. Is screening justified? Mil Med 2012;177(1):96–8. 9. Samamé J, Kaul A, Garza U, Echeverria A, Galvani C. Laparoscopic aneurysm resection and splenectomy for splenic artery aneurysm in the third trimester of pregnancy. Surg Endosc 2013;27(8):2988–91.

Splenic Artery Aneurysm in Pregnancy.

Contexte : L’anévrisme de l’artère splénique (AAS) est une maladie rare qui affecte principalement les femmes et dont la prise en charge pose des défi...
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