Journal of Perinatology (2012) 32, 147–149 r 2012 Nature America, Inc. All rights reserved. 0743-8346/12 www.nature.com/jp PERINATAL/NEONATAL CASE PRESENTATION Pseudoaneurysm formation after umbilical arterial catheterization: an uncommon but potentially life-threatening complication

MJ So1, D Kobayashi2, E Anthony1 and J Singh1 1Department of Radiology, Wake Forest University School of Medicine, Winston-Salem, NC, USA and 2Department of Pediatrics, Wake Forest University School of Medicine, Winston-Salem, NC, USA

at an outside institution. APGAR scores were 3 and 7 at 1 and Relatively straightforward placement of an umbilical arterial can be 5 min, respectively. The newborn appeared depressed, hypotonic, complicated by psuedoaneurysm formation in the presence of a pale and mottled; she had significant petechiae and bruising of the coagulopathy. We describe a case of a neonate, where bedside ultrasound skin. studies revealed an elevated prothrombin time had a key role in the timely diagnosis of this complication and prevented a (PT) and partial thromboplastin time (PTT), and a platelet count potentially fatal outcome. Pertinent imaging findings are described with a of 195 000 per mm3. A low-lying umbilical venous catheter (UVC) brief literature review. was placed. She was then transferred to our institution for further Journal of Perinatology (2012) 32, 147–149; doi:10.1038/jp.2011.93 management. Keywords: pseudoaneurysm; umbilical arterial catheterization; Upon admission, the UVC was replaced. Two attempts at UAC coagulopathy; abdominal ultrasound; hemoperitoneum placement using 3.5 Fr and 5.0 Fr Argyle (Tyco Healthcare Group LP, Mansfield, MA, USA) were unsuccessful. The lines threaded easily; however, no blood return was obtained. The UAC Introduction was removed and a peripheral arterial catheter was placed instead. Umbilical arterial catheter (UAC) placement is a Coagulation studies documented a PT of 20.8 s, PTT 65.1 s, procedure, commonly performed in neonates, to enable arterial international normalized ratio 2.15, fibrinogen 141 mg dlÀ1 and pressure monitoring, blood sampling and exchange transfusion. platelets 270 000 per mm3. Extensive coagulation work up Access is obtained through the umbilical , with the catheter revealed a negative direct Coomb’s test and normal for age normally following an inferior course until it enters the common coagulation factors (IX 27%, VIII 54%, XII 53% and XI 47%). iliac artery, where it turns and ascends into the aorta.1 Appropriate Shortly after the attempted UAC placement, the patient’s placement of the catheter tip is in the descending aorta, either hemoglobin dropped from 12 to 5.4 g dlÀ1, requiring aggressive between the T6 and T10 levels or at the level of L3 above the fluid resuscitation. Urgent abdominal ultrasound was performed aortic bifurcation.2,3 The complication rate of UAC is B10%4; due to the sudden drop of hemoglobin and the development of a complications include thromboembolism, mechanical vascular lower abdominal mass; it demonstrated a heterogeneous mass injury such as perforation, infection and pseudoaneurysm measuring 4.0 Â 2.7 Â 3.6 cm below the umbilicus, subjacent formation. In our case, abdominal ultrasound demonstrated the to the anterior abdominal wall and superior to the dome of the pseudoaneurysm formation caused by UAC placement; subsequent bladder. This mass demonstrated mixed echogenicity with a surgical resection of the pseudoaneurysm was successful. whorled pattern peripherally and more homogenous central echogenicity, with low level internal echoes and arterial flow on color Doppler (see Figure 1). The large amount of heterogeneously Case echogenic material surrounding the superior and left lateral A female newborn was born at 30-week gestation to a 19-year-old aspects of the lesion was concerning for thrombus. Anterior to the mother via C-section for placental abruption and fetal bradycardia, right aspect of the lesion, the right umbilical artery was identified. The left umbilical artery was identified anteriorly draped over the Correspondence: Dr J Singh, Department of Radiology, Wake Forest University School of hematoma along the abdominal wall, although no definite Medicine, Medical Center Boulevard, Winston-Salem, NC 27157, USA. E-mail: [email protected] communication could be demonstrated with the above described Received 27 December 2010; revised 14 April 2011; accepted 25 May 2011 mass. The lesion did extend extraperitoneally up to the level of the Pseudoaneurysm due to UAC MJ So et al 148

soft tissue and fascia, and they are considered an unstable, temporary containment of imminent exsanguination if associated with a major arterial vessel. As such, they are surgical emergencies that require prompt diagnosis. Pseudoaneurysm formation is an uncommon but potentially devastating complication of UAC placement. The use of UACs in neonates began in the 1960s, and our review of the literature found that a number of case reports exist from as early as the 1970s; however, these primarily describe pseudoaneurysms of the descending aorta2,6–10 or iliac artery.10,11 The majority of these pseudoaneurysms were mycotic in nature,2,5–11 as opposed to the index case, which appears post-traumatic given the time course and clinical history. Given the location of the hematoma, we believe the catheter trauma occurred at the level of the acutely, Figure 1 Frame capture of 2D duplex grayscale and color Doppler transverse axis sonogram of the anterior lower abdomen demonstrates vascular flow in a anteriorly angled take off of the umbilical artery from the internal portion of a large, heterogeneous mass. Note the anterior course of the umbilical iliac artery. This supposition was corroborated at surgery. in relation to the mass (white arrows). Several prior reports discuss the local trauma of a stiff catheter against the vascular wall, focal turbulent flow at the catheter tip and secondary inflammatory changes as contributing factors to junction of the left umbilical artery and the iliac artery. pseudoaneurysm formation.2,3,8,10,12 Hemophilia A, hemophilia The ultrasound conclusion reported the mass as B and von Willibrand’s disease have all been associated with an umbilical pseudoaneurysm with hemoperitoneum. traumatic/iatrogenic pseudoaneurysm formation.13,14 Pediatric surgery planned abdominal exploration. Before Coagulopathies of these types increase the risk of invasive surgery, she became pulseless and cardiopulmonary resuscitation procedures, but in our patient, these conditions were not present. was initiated, followed by emergent laparotomy. Dark hematoma Instead, there was only a transient nonspecific prolongation of filled both the intraperitoneal cavity and extraperitoneal spaces PT and PTT. There is no available literature linking these along the external iliac vessels and left inguinal region. The parameters to incidence of in this age group. abdomen was packed, and after pharmaceutical resuscitation with Mlekusch et al.15 found a 100% positive predictability between epinephrine and atropine, spontaneous cardiac activity and iatrogenic occurrence of psuedoaneurysm and circulation returned. Definitive central venous access was platelet count below 200 Â 109 per l. They also found that patients eventually secured and further exploration of the abdomen showed with pseudoaneurysms more frequently received an aggressive an adventitial hematoma of the left umbilical artery extending into antithrombotic regimen; intravenously administered abciximab the extraperitoneum to the level of the junction of the umbilical had a significant effect on the development of pseudoaneurysms and internal iliac arteries. The artery was ligated close to the and treatment with clopidogrel also was a risk factor. The study was iliac artery and the hematoma was evacuated. limited as the authors did not use any other coagulation Of note, the patient received platelet transfusions, vitamin K and parameters in the study. We feel that this subgroup would also have fresh frozen plasma perioperatively. Over the course of the deranged parameters like PT, PTT and activated clotting time. admission, the prolonged PT and PTT normalized. The Critical maintenance of coagulation parameters is standard coagulopathy resolved completely and was not felt to be hereditary practice while treating through an endovascular route.16 in nature. The patient did have bilateral grade III germinal matrix The role of sepsis has been linked to pseudoaneurysm and hemorrhages on postoperative day 1. This was complicated by coagulopathy.17 In this case, the patient was empirically started on hydrocephalus, which required staged ventriculoperitoneal shunt ampicillin and gentamicin due to a risk of sepsis. Blood cultures placement. At the time of this report, she is in good health and were negative on day of life 3 and thus while sepsis could not receives regular follow-up as an outpatient. entirely be excluded, its role in coagulopathy in this case cannot be definitely substantiated. We think that in our case, the occurrence of iatrogenic Discussion pseudoaneurysm is likely multifactorial. Although the platelet Pseudoaneurysms are contained hematomas that form at the site count in our case was within normal limits for age, transient of a complete rupture of a vessel wall, including the adventitial, coagulopathy may have contributed to pseudoaneurysm formation medial and intimal layers.5 Generally composed of blood and and hemoperitoneum. Alternatively, the whole picture may organizing thrombus, these lesions are bounded by surrounding represent a consumptive coagulopathy.18

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At least one case report of pseudoaneurysm formation following 6 Malloy MH, Nichols MM. False abdominal : an unusual complication UAC placement was published by Katz et al. in 1986.5 We found no of umbilical arterial catheterization for exchange transfusion. J Pediatr 1977; 90: report of associated hemoperitoneum. This complication may have 285–286. been more frequently encountered; however, the relative paucity of 7 Spangler JG, Kleinberg F, Fulton RE, Barnhorst DA, Ritter DG. False aneurysm of the descending aorta: a complication of umbilical artery catheterization. Am J Dis Child reported cases would suggest otherwise. Pseudoaneurysm formation 1977; 131: 1258–1259. should be recognized as an uncommon but serious iatrogenic 8 Wynn ML, Rowen M, Rucker RW, Sperling DR, Gazzaniga AB. Pseudoaneurysm of the complication of a relatively standard procedure in neonatal thoracic aorta: a late complication of umbilical artery catheterization. Annals Thorac intensive care. Care should be exercised when executing any Surg 1982; 34: 186–191. invasive procedure and a differential diagnosis of a traumatic 9 Saabye J, Elbkirk A, Smith C. Mycotic aneurysm of the thoracic aorta as a late complication of umbilical artery catheterization. Scand J Thor Cardiovasc Surg 1986; pseudoaneurysm should be considered when clinical signs of 20: 179–182. , decreasing hemoglobin or shock present immediately 10 Rabin E, Vye MV, Farrell EE. Umbilical artery catheterization complicated by after a difficult or unsuccessful catheterization. In the setting of multiple mycotic aortic aneurysms. Arch Pathol Lab Med 1986; 110: 442–444. new intraperitoneal fluid, generalized hemoperitoneum should be 11 Lally KP, Sherman NJ. Iliac artery pseudoaneurysm following umbilical artery suspected and operative intervention should be considered. catheterization. Surgery 1986; 101: 636–638. 12 Shah V, Hellmann J, Chait P, Connolly B. Radiology casebook. Pseudoaneurysm of the right internal iliac artery after umbilical artery catheterization: case report and review of the literature. J Perinatol 2000; 20: 392–396. Conflict of interest 13 Kumar R, Pruthi RK, Kobrinsky N, Shaughnessy WJ, McKusick MA, Rodriguez V. The authors declare no conflict of interest. Pelvic pseudotumor and pseudoaneurysm in a pediatric patient with moderate hemophilia B: successful management with arterial embolization and surgical excision. Pediatr Blood Cancer 2011; 56: 484–487. References 14 Saarela MS, Tiitola M, Lappalainen K, Vikatmaa P, Pinoma¨ki A, Alberty A et al. Pseudoaneurysm in association with a knee endoprothesis operation in an inhibitor- 1 Schlesinger AE, Braverman RM, DiPietro MA. neonates and umbilical venous catheters: positive haemophilia A patientFtreatment with local . Haemophilia 2010; normal appearance, anomalous positions, complications, and potential aid to 6: 686–688. diagnosis. AJR 2003; 180: 1147–1153. 15 Mlekusch W, Haumer M, Mlekusch I, Dick P, Steiner-Boeker S, Bartok A et al. 2 Bapat VN, Dinesh RS, Dhaded SB, Khandeparkar JMS, Borwankar SS, Magotra RA. Prediction of iatrogenic pseudoaneurysm after percutaneous endovascular procedures. Delayed presentation of false abdominal aortic aneurysm following umbilical artery Radiology 2006; 240: 597–602. catheterization. Thorac cardiovasc Surg 1997; 45: 154–157. 16 Song JK, Niimi Y, Fernandez PM, Brisman JL, Buciuc R, Kupersmith MJ et al. 3 Green C, Yohannon MD. Umbilical arterial and venous catheters: placement, use, and Thrombus formation during coil placement: treatment with complications. Neonatal Netw 1998; 17: 23–28. intra-arterial abciximab. AJNR Am J Neuroradiol 2004; 25: 1147–1153. 4 Cribari C, Meadors FA, Crawford ES, Coselli JS, Safi HJ, Svensson LG et al. 17 Wyers MR, McAlister WH. Umbilical artery catheter use complicated by pseudoaneurysm Thoracoabdominal aortic aneurysm associated with umbilical artery catheterization: of the aorta. Pediatr Radiol 2002; 32: 199–201. case report and review of the literature. J Vasc Surg 1992; 16: 75–86. 18 Yuan SM, Shinfeld A, Tager S, Raanani E. Aortic aneurysm-induced disseminated 5 Katz ME, Perlman JM, Tack ED, McAlister WH. Neonatal umbilical artery intravascular coagulopathy: successful surgical repair. Vascular 2009; 17: psaeudoaneurysm: sonographic evaluation (Case report). AJR 1986; 147: 322–324. 55–59.

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