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Letters to the Editor

Yoon et al



JVIR

We were asked to place an IVC filter in a 46-year-old woman who presented with progression of deep vein thrombosis to symptomatic pulmonary embolism despite therapeutic anticoagulation. A commercially available Bard Meridian retrievable IVC filter was selected. These filters have shown prolonged retrieval capability based on similar designs from the same manufacturer (3). We implanted the filter via a right common femoral vein approach, with an approximate 201 tilt (Fig, a). Three months after placement, upper abdominal images from a chest computed tomography (CT) examination showed an intact filter with all 12 limbs attached. Because the patient’s deep vein thrombosis burden decreased with anticoagulation and the CT scan showed no residual pulmonary embolism, we scheduled filter removal 2 weeks later. Initial imaging during the retrieval attempt displayed a fractured arm of the filter attached to the IVC wall (Fig, b). This was a surprise given that the CT scan performed a few weeks earlier had shown an intact filter. We removed the major component of the filter by using an 11-F goose neck–type snare from a vena cava filter retrieval system (Cook, Bloomington, Indiana); this component remained intact during the removal. The intravascular fragment adherent to the IVC was retrieved by using a 9–15-mm trilobed snare (EnSnare; Merit Medical, South Jordan, Utah). Inspection of the major filter component showed that, in fact, an additional arm was missing. On a CT angiogram of the chest, this second fractured arm was located within the right ventricle, partially penetrating the wall (Fig, c). The patient did not have any chest pain, arrhythmia, or shortness of breath related to this fragment. Retrieval of the intraventricular filter limb was performed the next day. We placed a curved-tip 10-F, 38.5cm Flexor CheckFlo II Introducer sheath (Cook) into the right atrium for guidance. A transesophageal echocardiogram probe was placed to visualize the pericardial sac for potential bleeding. We chose an Amplatz GooseNeck 25-mm snare with catheter (ev3, Plymouth, Minnesota) to retrieve the fragment. The filter fragment was removed with mild tactile resistance, and there were minimal premature ventricular contractions during the

procedure (Fig, d). A ventriculogram immediately after removal showed no active extravasation. Subsequent CT angiograms of the chest showed no myocardial or pericardial bleeding. The patient was asymptomatic on follow-up consultation 2 months later. Regardless of cause, embolization of fractured IVC filter fragments has been associated with severe complications, including ventricular tachycardia, cardiac perforation, and death (1,2). High retrieval rates of the nonfractured component have been demonstrated, and fractured limbs have been successfully removed approximately 50% of the time without significant complication (4). However, arrhythmia, development of pericardial effusion, or valve or cordae tendineae injury are all potential risks from retrieval of limbs embolized to the heart. The present case presents spontaneous fracture of two limbs of a Bard Meridian IVC filter, with one fragment embolizing to the heart. It is plausible that the tilted position following placement contributed to the filter fracture. However, it is important to note that CT imaging 3 months after placement showed an intact filter; both limbs fractured, in the absence of any manipulation, at the apex point where they are glued together. Given the findings of this case report, and the history of previous filters from this manufacturer resulting in similar complications, we believe the fracture potential of the Meridian filter should be further evaluated.

Transarterial Embolization of a Cervicofacial Hemangioma Associated with Kasabach–Merritt Syndrome in a Premature Neonate

Hyun Kyung Park, MD Hey Suk Yun, MD Hyun Ju Lee, MD Departments of Radiology (K.Y., Y.-J.L., D.W.P.) and Pediatrics (H.K.P., H.S.Y., H.J.L.) Hanyang University College of Medicine Hanyang University Hospital 17 Haendang-Dong Sungdong-Ku Seoul 133-792, Korea

From: Kibo Yoon, MD Young-Jun Lee, MD Dong Woo Park, MD

None of the authors have identified a conflict of interest. http://dx.doi.org/10.1016/j.jvir.2013.08.026

REFERENCES 1. Tam MD, Spain J, Lieber M, Geisinger M, Sands MJ, Wang W. Fracture and distant migration of the Bard Recovery filter: a retrospective review of 363 implantations for potentially life-threatening complications. J Vasc Interv Radiol 2012; 23:199–205. 2. Nicholson W, Nicholson WJ, Tolerico P, et al. Prevalence of fracture and fragment embolization of Bard retrievable vena cava filters and clinical implications including cardiac perforation and tamponade. Arch Intern Med 2010; 170:1827–1831. 3. Binkert CA, Drooz AT, Caridi JG, et al. Technical success and safety of retrieval of the G2 filter in a prospective, multicenter study. J Vasc Interv Radiol 2009; 20:1449–1453. 4. Vijay K, Hughes JA, Burdette AS, et al. Fractured Bard Recovery, G2, and G2 Express inferior vena cava filters: incidence, clinical consequences, and outcomes of removal attempts. J Vasc Interv Radiol 2012; 23:188–194.

Editor: Here we describe successful transarterial embolization in a prematurely delivered neonate with Kasabach–Merritt

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Number 12



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2013

Figure 1. Photograph shows reddish-colored mass covering the whole of right side of the face and neck.

Figure 2. Axial fluid-attenuated inversion recovery MR image shows a large mass in the right face with deep infiltration to masticator spaces and intracranial hematoma in the left frontotemporal region.

syndrome (KMS) associated with cervicofacial hemangioma and a large intracranial hematoma. A male infant was delivered by cesarean section at 33 weeks of gestation because of an intracranial hematoma noted on fetal ultrasonography (US). His birth weight was 2.17 kg, and 5-minute Apgar score was 5. He had a reddishcolored mass covering the whole of the right side of his face and neck (Fig 1). Initial laboratory tests revealed thrombocytopenia (o 10  103/mm3; normal range,

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150–450  103/mm3), prolonged prothrombin time (13.8 s; normal range, 9.0–13.2 s), prolonged activated partial thromboplastin time (85 s; normal range, 25–39 s), increased International Normalized Ratio (1.29; normal range, 0.82–1.2), increased fibrin degradation products (4 20 mg/mL; normal range, 0–5 mg/mL), decreased fibrinogen (161 mg/dL; normal range, 190– 430 mg/dL), and increased D-dimer (3.44 mg/L; normal range, 0–0.24 mg/L). Increased vascularity of the mass was evident on US. With a presumptive diagnosis of KMS caused by a vascular mass, conservative management was initiated with corticosteroids (prednisolone 2 mg/kg/d), platelet concentrates (320 mL/d), and fresh frozen plasma (320 mL/d). Magnetic resonance (MR) imaging 2 days after birth showed a hypervascular mass in the right cervicofacial region with deep infiltration into masticator spaces (Fig 2). Hematologic parameters did not normalize with medical treatment, and a significant risk of severe intracranial hemorrhage was present. Embolization of the mass was therefore attempted on day 4 after delivery. Under general anesthesia, a 4-F sheath was inserted percutaneously in the right femoral artery. An angiogram with a 4-F angiocatheter positioned at the right common carotid artery showed extensive contrast staining of the mass, fed by hypertrophied internal maxillary and facial arteries, without evidence of arteriovenous shunting (Fig 3a). In a coaxial manner, a microcatheter (Prowler Plus; Codman and Shurtleff, Raynham, Massachusetts) was advanced through the 4-F catheter into various feeder vessels, and embolization was performed with 250–355-mm polyvinyl alcohol particles. Final angiogram showed significantly reduced contrast staining compared with the initial angiogram (Fig 3b). The use of any fluid materials, including saline flushing and contrast media, was restricted to as small a volume as possible to prevent fluid overload. The following day, coagulation parameters were within normal ranges (prothrombin time, 12.3 s; International Normalized Ratio, 1.14; activated partial thromboplastin time, 39 s). Platelet count increased gradually and stabilized at approximately 100  103 from day 3 after embolization. The mass shrank progressively during the patient’s remaining days in the hospital, and his general condition also stabilized. A punch biopsy was performed on day 14 after embolization, and the mass was confirmed as infantile capillary hemangioma. Followup MR imaging on day 18 after embolization showed reduced size of the mass without aggravation of the intracranial hematoma. The infant was discharged during the fifth week after embolization without any signs of skin necrosis or cranial nerve palsy. Follow-up MR imaging at week 22 after embolization showed significantly reduced size of hemangioma and encephalomalacia from previous intracranial hematoma in the left frontotemporal region (Fig 4). Treatment with corticosteroids on an outpatient basis was continued,

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Letters to the Editor

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JVIR

Figure 3. (a) Angiography before embolization through right common carotid artery shows hypertrophied branches of right extracranial artery with contrast staining of cervicofacial hemangioma. (b) Angiography after embolization through right internal maxillary artery shows reduced staining of cervicofacial hemangioma.

Figure 4. Follow-up fluid-attenuated inversion recovery MR image during the 22nd week after embolization shows markedly reduced size of hemangioma and intracranial hematoma.

with gradual tapering, and the patient remained in clinically stable condition at 6-month follow-up. KMS was first described by Kasabach and Merritt in 1940 (1) and refers to a condition with thrombocytopenia and consumptive coagulopathy associated with a vascular tumor. Although there are many treatment options for KMS, including steroids, interferon therapy, chemotherapy, radiation therapy, surgery, and embolization, treatment guidelines are not fully established, and multimodal treatments have been used with varying results (2). There are other articles describing the use of trans-

arterial embolization for treatment of neonatal KMS associated with vascular tumors involving not head and neck but extremity and abdominal lesions (3,4). As in the case of cervicofacial vascular tumors, transarterial embolization carries risks of cranial nerve injury and accidental intracranial embolism. In addition, endovascular treatment in the preterm infant is challenging because of the dangers arising from general anesthesia, fluid overload, and technical difficulties associated with tiny vessels in a tiny individual. The case described here shows that transarterial embolization is technically feasible even for a premature infant and has an immediate impact on hematologic parameters. Polyvinyl alcohol particles 250–355 mm in size were used safely for embolization of the tumor fed by the external carotid artery, without unwanted complications. Careful restriction of fluid input during the procedure can prevent volume overload. Although embolization alone may not be sufficient to achieve regression of the whole tumor, it provides time to wait for any effect of the medical treatment or for spontaneous involution. Transarterial embolization is feasible, safe, and effective in premature infants.

REFERENCES 1. Kasabach HH, Merritt KK. Capillary hemangioma with extensive purpura. Report of a case. Am J Dis Child 1940; 59:1063–1070. 2. Hall GW. Kasabach–Merritt syndrome: pathogenesis and management. Br J Haematol 2001; 112:851–862. 3. Garcia-Monaco R, Giachetti A, Peralta O, et al. Kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon: successful treatment with embolization and vincristine in two newborns. J Vasc Interv Radiol 2012; 23:417–422. 4. Zhou SY, Li HB, Mao YM, Liu PY, Zhang J. Successful treatment of Kasabach–Merritt syndrome with transarterial embolization and corticosteroids. J Pediatr Surg 2013; 48:673–676.

Transarterial embolization of a cervicofacial hemangioma associated with Kasabach-Merritt syndrome in a premature neonate.

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