Letter to the Editor
Superior vena cava syndrome as a complication of intravenous immunoglobulin treatment
Síndrome de vena cava superior como complicación del tratamiento con inmunoglobulinas intravenosas
J.A. Crespo-Burillo
, R. Alarcia-Alejos, J.L. Capablo-Liesa
Corresponding author
Servicio de Neurología, Hospital Universitario Miguel Servet, Zaragoza, Spain
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Crespo-Burillo, R. Alarcia-Alejos, J.L. Capablo-Liesa" "autores" => array:3 [ 0 => array:4 [ "nombre" => "J.A." "apellidos" => "Crespo-Burillo" "email" => array:1 [ 0 => "josanjoseli@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "R." "apellidos" => "Alarcia-Alejos" ] 2 => array:2 [ "nombre" => "J.L." "apellidos" => "Capablo-Liesa" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Neurología, Hospital Universitario Miguel Servet, Zaragoza, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de vena cava superior como complicación del tratamiento con inmunoglobulinas intravenosas" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1023 "Ancho" => 975 "Tamanyo" => 61606 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Contrast chest CT scan (multiplanar reconstruction). The catheter (asterisk) occupies almost the entire lumen of the proximal segment of the superior vena cava (arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Intravenous immunoglobulins are a frequent treatment for a variety of neurological diseases.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> This treatment has been associated with thromboembolic complications, with an incidence ranging between 1.2% and 11.3%.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The pathogenic mechanisms of the disease include increased plasma viscosity, increased platelet count and adhesion, and presence of procoagulant antibodies and coagulation factors not eliminated by immunoglobulin fractionation.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Superior vena cava thrombosis in patients receiving intravenous immunoglobulins is an infrequent complication that has rarely been described in the literature.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Management of this complication may be difficult, especially in patients with a central venous catheter, since no specific management guidelines have been established to date.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">We present the case of a 57-year-old female smoker diagnosed with chronic inflammatory demyelinating polyneuropathy. She was in treatment with azathioprine, deflazacort 6<span class="elsevierStyleHsp" style=""></span>mg/24<span class="elsevierStyleHsp" style=""></span>h, and intravenous immunoglobulins dosed at 0.5<span class="elsevierStyleHsp" style=""></span>g/kg/day and administered over 12 hours per day for 4 days every month. She had no other relevant history. Immunglobulin therapy had been started 3 years earlier, by means of a subcutaneous reservoir attached to a catheter in the superior vena cava. The catheter had been implanted following multiple episodes of thrombophlebitis and extravasation. She had a 3-month history of cervico-facial oedema. A metabolic study performed to rule out alterations linked to oedema yielded normal results, including normal thyroid and renal function, normal albumin and total protein levels in serum, normal protein levels in urine, and no changes in urine sediment. A transthoracic echocardiogram displayed no abnormalities. A chest CT scan with contrast revealed that the catheter occupied nearly the entire lumen of the proximal segment of the superior vena cava (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A cavography demonstrated mild stenosis of the superior vena cava, which was complicated by a thrombus around the catheter (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). A complete autoimmunity and hypercoagulation study ruled out a prothrombotic state. We decided to start anticoagulation therapy with enoxaparin dosed at 60<span class="elsevierStyleHsp" style=""></span>mg/12<span class="elsevierStyleHsp" style=""></span>h. The catheter was removed a month later. A follow-up cavography revealed no thrombosis in the lumen of the vena cava or around the catheter; mild residual stenosis persisted. Cervico-facial oedema disappeared. We decided to continue treatment with peripheral venous immunoglobulins, maintain anticoagulation therapy with acenocoumarol for an additional 5 months, and subsequently administer antithrombotics with each infusion (enoxaparin 40<span class="elsevierStyleHsp" style=""></span>mg/24<span class="elsevierStyleHsp" style=""></span>h) as prophylaxis.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Superior vena cava syndrome results from obstruction of blood flow through the superior vena cava due to compression or occlusion. In around 60% of the cases, this syndrome is caused by malignancies, the most frequent being lung carcinoma and lymphoma. The most common benign aetiology has to do with placement of such intravascular devices as reservoir catheters or pacemaker electrodes<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a>; the associated incidence of thrombosis varies from study to study (2%-67%).<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Venous thrombosis is caused by 3 main factors known collectively as the Virchow triad: stasis, hypercoagulability, and endothelial injury.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> Central venous catheters may damage the vascular endothelium and promote venous stasis by obstructing blood flow through the vena cava. Patients with these catheters may present hypercoagulable states associated with the underlying disease, with primary clotting disorders, or with the treatment itself. In our patient, immunoglobulin treatment may have favoured this mechanism by completing the triad.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Risk of thromboembolism in patients receiving immunoglobulins must be calculated based on age, presence of cardiovascular risk factors, immobility, prior thromboembolic complications, and concomitant use of prothrombotic drugs, including corticosteroids, which favour platelet aggregation and inhibit the fibrinolytic system. The literature reports cases of deep vein thrombosis secondary to immunoglobulin use, normally at high doses or after long treatment periods.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,8</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Given the lack of randomised clinical trials, management of superior vena cava syndrome secondary to thrombosis of an intravascular catheter has not been protocolised.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Treatment must therefore be tailored to each patient according to the degree of stenosis and severity of clinical symptoms. Less severe cases, such as that of our patient, may benefit from conservative treatment with anticoagulants until symptoms resolve.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> We opted to remove the reservoir in our patient since it was believed to significantly increase the risk of a new thrombotic event. Given that early catheter removal has failed to deliver a better prognosis in thrombosis,<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> we decided to remove the catheter a month after initiating anticoagulation therapy to prevent potential thromboembolic complications secondary to thrombus mobilisation. We maintained anticoagulation therapy for 6 months since the risk factor for deep vein thrombosis (the catheter) was understood to be temporary.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> In more severe cases, endovascular treatment with balloon angioplasty and stenting is the first-line option since its effectiveness is similar to that of surgery and it is associated with fewer complications.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> The duration of anticoagulation therapy for patients in whom the catheter remains in place has not been established.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> We recommend applying preventive measures before immunoglobulin infusion (hydration, antiplatelet drugs or low molecular weight heparins, infusion during no less than 8 hours, and administering the normal dose [2<span class="elsevierStyleHsp" style=""></span>g/kg] in fractions of 0.4<span class="elsevierStyleHsp" style=""></span>g/kg/day for 5 days) given the high risk of thrombosis, even in patients with no history of thromboembolic events.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3,5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion, presence of a central venous catheter may promote the thrombogenic mechanisms associated with immunoglobulin treatment and result in superior vena cava syndrome secondary to superior vena cava thrombosis. When this route of treatment must be used, antithrombotic measures should be taken before each infusion.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Crespo-Burillo JA, Alarcia-Alejos R, Capablo-Liesa JL. Síndrome de vena cava superior como complicación del tratamiento con inmunoglobulinas intravenosas. Neurología. 2017;32:202–204.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1023 "Ancho" => 975 "Tamanyo" => 61606 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Contrast chest CT scan (multiplanar reconstruction). The catheter (asterisk) occupies almost the entire lumen of the proximal segment of the superior vena cava (arrow).</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1233 "Ancho" => 900 "Tamanyo" => 73840 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cavography displaying mild stenosis of the proximal segment of the superior vena cava, complicated by a thrombus around the catheter (arrow).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The use of intravenous immunoglobulins in neurology" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "T. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 7 | 1 | 8 |
| 2024 September | 18 | 7 | 25 |
| 2024 August | 30 | 15 | 45 |
| 2024 July | 19 | 8 | 27 |
| 2024 June | 9 | 5 | 14 |
| 2024 May | 19 | 5 | 24 |
| 2024 April | 18 | 8 | 26 |
| 2024 March | 33 | 10 | 43 |
| 2024 February | 14 | 3 | 17 |
| 2024 January | 18 | 12 | 30 |
| 2023 December | 28 | 5 | 33 |
| 2023 November | 40 | 3 | 43 |
| 2023 October | 46 | 6 | 52 |
| 2023 September | 24 | 6 | 30 |
| 2023 August | 17 | 2 | 19 |
| 2023 July | 18 | 6 | 24 |
| 2023 June | 23 | 6 | 29 |
| 2023 May | 32 | 9 | 41 |
| 2023 April | 32 | 0 | 32 |
| 2023 March | 50 | 5 | 55 |
| 2023 February | 17 | 8 | 25 |
| 2023 January | 35 | 6 | 41 |
| 2022 December | 39 | 6 | 45 |
| 2022 November | 39 | 14 | 53 |
| 2022 October | 23 | 9 | 32 |
| 2022 September | 33 | 16 | 49 |
| 2022 August | 20 | 18 | 38 |
| 2022 July | 19 | 12 | 31 |
| 2022 June | 12 | 7 | 19 |
| 2022 May | 14 | 15 | 29 |
| 2022 April | 14 | 11 | 25 |
| 2022 March | 16 | 13 | 29 |
| 2022 February | 31 | 6 | 37 |
| 2022 January | 29 | 7 | 36 |
| 2021 December | 31 | 10 | 41 |
| 2021 November | 35 | 11 | 46 |
| 2021 October | 54 | 12 | 66 |
| 2021 September | 40 | 14 | 54 |
| 2021 August | 40 | 13 | 53 |
| 2021 July | 27 | 5 | 32 |
| 2021 June | 33 | 12 | 45 |
| 2021 May | 52 | 17 | 69 |
| 2021 April | 99 | 24 | 123 |
| 2021 March | 69 | 11 | 80 |
| 2021 February | 52 | 9 | 61 |
| 2021 January | 51 | 10 | 61 |
| 2020 December | 30 | 12 | 42 |
| 2020 November | 36 | 6 | 42 |
| 2020 October | 45 | 9 | 54 |
| 2020 September | 35 | 14 | 49 |
| 2020 August | 30 | 5 | 35 |
| 2020 July | 11 | 4 | 15 |
| 2020 June | 35 | 9 | 44 |
| 2020 May | 31 | 15 | 46 |
| 2020 April | 25 | 7 | 32 |
| 2020 March | 39 | 3 | 42 |
| 2020 February | 31 | 4 | 35 |
| 2020 January | 31 | 1 | 32 |
| 2019 December | 40 | 7 | 47 |
| 2019 November | 23 | 5 | 28 |
| 2019 October | 24 | 2 | 26 |
| 2019 September | 29 | 4 | 33 |
| 2019 August | 21 | 0 | 21 |
| 2019 July | 23 | 10 | 33 |
| 2019 June | 47 | 25 | 72 |
| 2019 May | 123 | 11 | 134 |
| 2019 April | 56 | 7 | 63 |
| 2019 March | 14 | 4 | 18 |
| 2019 February | 9 | 8 | 17 |
| 2019 January | 6 | 4 | 10 |
| 2018 December | 5 | 1 | 6 |
| 2018 November | 6 | 4 | 10 |
| 2018 October | 18 | 4 | 22 |
| 2018 September | 24 | 0 | 24 |
| 2018 August | 4 | 0 | 4 |
| 2018 July | 2 | 3 | 5 |
| 2018 June | 2 | 2 | 4 |
| 2018 May | 6 | 2 | 8 |
| 2018 April | 6 | 1 | 7 |
| 2018 March | 4 | 0 | 4 |
| 2018 February | 5 | 1 | 6 |
| 2018 January | 4 | 4 | 8 |
| 2017 December | 5 | 1 | 6 |
| 2017 November | 7 | 0 | 7 |
| 2017 October | 9 | 1 | 10 |
| 2017 September | 8 | 3 | 11 |
| 2017 August | 16 | 1 | 17 |
| 2017 July | 13 | 0 | 13 |
| 2017 June | 10 | 4 | 14 |
| 2017 May | 23 | 5 | 28 |
| 2017 April | 19 | 3 | 22 |
| 2017 March | 3 | 5 | 8 |
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