Letter to the Editor
Postural orthostatic tachycardia syndrome, neurally mediated syncope, and joint hypermobility: a case report
Síndrome de taquicardia postural ortostática, síncopes autonomomediados e hiperlaxitud articular: a propósito de un caso
K. Berganzo
, B. Tijero, J.J. Zarranz, J.C. Gómez-Esteban
Corresponding author
Unidad de Disautonomía y Trastornos del Movimiento, Departamento de Neurociencias, Biocruces Health Research Institute, Universidad del País Vasco, Barakaldo, Spain
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Berganzo, B. Tijero, J.J. Zarranz, J.C. Gómez-Esteban" "autores" => array:4 [ 0 => array:4 [ "nombre" => "K." "apellidos" => "Berganzo" "email" => array:1 [ 0 => "koldo.berganzocorrales@osakidetza.net" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "B." "apellidos" => "Tijero" ] 2 => array:2 [ "nombre" => "J.J." "apellidos" => "Zarranz" ] 3 => array:2 [ "nombre" => "J.C." "apellidos" => "Gómez-Esteban" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Unidad de Disautonomía y Trastornos del Movimiento, Departamento de Neurociencias, Biocruces Health Research Institute, Universidad del País Vasco, Barakaldo, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de taquicardia postural ortostática, síncopes autonomomediados e hiperlaxitud articular: a propósito de un caso" ] ] "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" => 665 "Ancho" => 1034 "Tamanyo" => 79274 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Signs of joint hypermobility in the physical examination.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of an adolescent girl aged 17 with no relevant medical history who was assessed in our epilepsy unit due to a 2-year history of repeated syncope, preceded by pallor and discomfort. There were no abnormal movements during episodes and the patient recovered in minutes with no subsequent confusion or loss of sphincter control. Episode frequency was variable with a maximum of 2 per week. Some occurred during physical exertion. She sometimes reported palpitations while standing or walking. She had also experienced repeated sprains.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was examined by the cardiology and neurology departments; she had visited the emergency department on 14 previous occasions and been admitted 6 times, including admission to the intensive care unit once due to suspected epilepsy. At that time, a differential diagnosis was performed for psychogenic seizures and epilepsy. The patient was treated with 2<span class="elsevierStyleHsp" style=""></span>g levetiracetam/24<span class="elsevierStyleHsp" style=""></span>hours with no signs of improvement. Test results were normal, except for isolated sinus tachycardia that was detected with an implantable loop recorder, and left-sided hippocampal malrotation detected by brain MRI. It was on this basis that doctors suspected epilepsy.</p><p id="par0015" class="elsevierStylePara elsevierViewall">The examination identified venous pooling in the lower limbs while standing, keloid where the implantable loop recorder had been placed, and joint hypermobility with a score of 5/9 on the Beighton scale (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient met Brighton diagnostic criteria for joint hypermobility (1 major and 2 minor criteria).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Doctors opted for a tilt table test and non-invasive haemodynamic monitoring with a Task Force<span class="elsevierStyleSup">®</span> Monitor (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). We observed low blood pressure (BP) at baseline and postural tachycardia with a heart rate above 120<span class="elsevierStyleHsp" style=""></span>bpm and cardioinhibitory syncope associated with vasodepressor response and a drop in peripheral resistance, together with an increase in stroke volume. All findings were compatible with neurally mediated syncope. Doctors began treatment with fludrocortisone dosed at 0.1<span class="elsevierStyleHsp" style=""></span>mg/day and non-pharmacological therapy; syncope episodes ceased completely.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Postural tachycardia syndrome has an estimated prevalence of 170 per 100<span class="elsevierStyleHsp" style=""></span>000 individuals. It is more common in women (5:1) and at ages between 20 and 40; aetiopathogenesis is heterogeneous. The condition essentially amounts to a variable degree of intolerance to orthostasis with the addition of symptoms secondary to hypoperfusion, such as difficulty concentrating, neck or thoracic pain due to tissue hypoperfusion, and symptoms of sympathetic hyperactivity with palpitations or tremor.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> This syndrome is closely linked to repeated episodes of neurally mediated syncope.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Diagnostic criteria are heart rate increase of 30<span class="elsevierStyleHsp" style=""></span>bpm (>40<span class="elsevierStyleHsp" style=""></span>bpm in patients aged 12 to 19 years) when standing or walking or on a tilt table without orthostatic hypotension, or heart rate above 120<span class="elsevierStyleHsp" style=""></span>bpm with no baseline arterial hypotension.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Non-invasive haemodynamic monitoring associated with BP monitoring over 24<span class="elsevierStyleHsp" style=""></span>hours is very useful for evaluating the condition.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Physical examination frequently reveals oedema and venous pooling in the lower limbs resulting from vasoconstriction disorders.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Type 3 Ehlers–Danlos syndrome (hypermobility syndrome) is related to postural tachycardia syndrome.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> It is characterised by presence of joint hypermobility (although this sign may be absent), skin abnormalities, and other signs.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Diagnosis is clinical; doctors use the Beighton score<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> and the Brighton diagnostic criteria<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> and rule out other potential entities.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Treatments may be pharmacological or non-pharmacological (increasing fluid and sodium intake, posture therapy, and cognitive behavioural therapy). Pharmacological treatments include 2 major groups. The first is typically used in cases with low baseline BP (fludrocortisone<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> and midodrine<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a>). The second is for patients with normal BP or arterial hypertension (beta-blockers and pyridostigmine).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> Drugs from both of the above groups may be used in combination on a case-by-case basis.</p><p id="par0045" class="elsevierStylePara elsevierViewall">These disorders are prevalent, underdiagnosed, and have a major impact on quality of life. However, patients may benefit from correct diagnostic and therapeutic management provided by specialists, and therefore avoid iatrogenic effects of testing and treatments and periodic visits to doctors and the emergency department. As a screening method, patients or their family members may measure BP and heart rate in the decubitus and standing positions. Proper management of these patients will require additional training for neurologists and the creation of specialised units.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Berganzo K, Tijero B, Zarranz JJ, Gómez-Esteban JC. Síndrome de taquicardia postural ortostática, síncopes autonomomediados e hiperlaxitud articular: a propósito de un caso. Neurología. 2014;29:447–449.</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" => 665 "Ancho" => 1034 "Tamanyo" => 79274 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Signs of joint hypermobility in the physical examination.</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" => 2239 "Ancho" => 3294 "Tamanyo" => 660525 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Non-invasive haemodynamic study with the Task Force<span class="elsevierStyleSup">®</span> Monitor during the tilt table test. (A) Postural tachycardia. (B) Syncope with initial cardioinhibitory response and vasodepressor response.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:11 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Postural tachycardia syndrome (POTS)" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "P.A. Low" 1 => "P. Sandroni" 2 => "M. Joyner" 3 => "W.K. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 5 | 0 | 5 |
| 2024 October | 33 | 10 | 43 |
| 2024 September | 70 | 7 | 77 |
| 2024 August | 34 | 7 | 41 |
| 2024 July | 41 | 6 | 47 |
| 2024 June | 42 | 8 | 50 |
| 2024 May | 45 | 7 | 52 |
| 2024 April | 22 | 8 | 30 |
| 2024 March | 39 | 5 | 44 |
| 2024 February | 32 | 4 | 36 |
| 2024 January | 71 | 2 | 73 |
| 2023 December | 50 | 6 | 56 |
| 2023 November | 61 | 9 | 70 |
| 2023 October | 58 | 14 | 72 |
| 2023 September | 41 | 4 | 45 |
| 2023 August | 45 | 7 | 52 |
| 2023 July | 50 | 10 | 60 |
| 2023 June | 43 | 5 | 48 |
| 2023 May | 48 | 2 | 50 |
| 2023 April | 52 | 1 | 53 |
| 2023 March | 67 | 15 | 82 |
| 2023 February | 40 | 1 | 41 |
| 2023 January | 52 | 4 | 56 |
| 2022 December | 45 | 7 | 52 |
| 2022 November | 40 | 8 | 48 |
| 2022 October | 37 | 36 | 73 |
| 2022 September | 36 | 14 | 50 |
| 2022 August | 36 | 11 | 47 |
| 2022 July | 33 | 9 | 42 |
| 2022 June | 25 | 7 | 32 |
| 2022 May | 31 | 11 | 42 |
| 2022 April | 54 | 12 | 66 |
| 2022 March | 53 | 8 | 61 |
| 2022 February | 63 | 6 | 69 |
| 2022 January | 49 | 9 | 58 |
| 2021 December | 24 | 12 | 36 |
| 2021 November | 29 | 9 | 38 |
| 2021 October | 31 | 13 | 44 |
| 2021 September | 21 | 13 | 34 |
| 2021 August | 27 | 6 | 33 |
| 2021 July | 26 | 7 | 33 |
| 2021 June | 29 | 8 | 37 |
| 2021 May | 41 | 14 | 55 |
| 2021 April | 84 | 38 | 122 |
| 2021 March | 59 | 9 | 68 |
| 2021 February | 24 | 10 | 34 |
| 2021 January | 47 | 11 | 58 |
| 2020 December | 38 | 7 | 45 |
| 2020 November | 28 | 14 | 42 |
| 2020 October | 31 | 9 | 40 |
| 2020 September | 25 | 12 | 37 |
| 2020 August | 30 | 11 | 41 |
| 2020 July | 20 | 16 | 36 |
| 2020 June | 36 | 8 | 44 |
| 2020 May | 42 | 17 | 59 |
| 2020 April | 22 | 3 | 25 |
| 2020 March | 37 | 11 | 48 |
| 2020 February | 22 | 5 | 27 |
| 2020 January | 38 | 7 | 45 |
| 2019 December | 38 | 17 | 55 |
| 2019 November | 21 | 10 | 31 |
| 2019 October | 24 | 20 | 44 |
| 2019 September | 37 | 103 | 140 |
| 2019 August | 21 | 106 | 127 |
| 2019 July | 55 | 89 | 144 |
| 2019 June | 46 | 111 | 157 |
| 2019 May | 104 | 99 | 203 |
| 2019 April | 54 | 111 | 165 |
| 2019 March | 12 | 63 | 75 |
| 2019 February | 29 | 10 | 39 |
| 2019 January | 18 | 12 | 30 |
| 2018 December | 17 | 18 | 35 |
| 2018 November | 22 | 1 | 23 |
| 2018 October | 30 | 7 | 37 |
| 2018 September | 17 | 10 | 27 |
| 2018 August | 9 | 3 | 12 |
| 2018 July | 13 | 6 | 19 |
| 2018 June | 10 | 3 | 13 |
| 2018 May | 17 | 7 | 24 |
| 2018 April | 13 | 2 | 15 |
| 2018 March | 15 | 3 | 18 |
| 2018 February | 6 | 5 | 11 |
| 2018 January | 6 | 6 | 12 |
| 2017 December | 17 | 5 | 22 |
| 2017 November | 12 | 14 | 26 |
| 2017 October | 16 | 4 | 20 |
| 2017 September | 13 | 9 | 22 |
| 2017 August | 20 | 9 | 29 |
| 2017 July | 17 | 14 | 31 |
| 2017 June | 22 | 9 | 31 |
| 2017 May | 26 | 9 | 35 |
| 2017 April | 18 | 9 | 27 |
| 2017 March | 15 | 22 | 37 |
| 2017 February | 33 | 7 | 40 |
| 2017 January | 17 | 6 | 23 |
| 2016 December | 29 | 13 | 42 |
| 2016 November | 24 | 14 | 38 |
| 2016 October | 41 | 16 | 57 |
| 2016 September | 92 | 11 | 103 |
| 2016 August | 62 | 8 | 70 |
| 2016 July | 12 | 5 | 17 |
| 2016 June | 32 | 10 | 42 |
| 2016 May | 36 | 32 | 68 |
| 2016 April | 26 | 34 | 60 |
| 2016 March | 30 | 62 | 92 |
| 2016 February | 29 | 16 | 45 |
| 2016 January | 22 | 17 | 39 |
| 2015 December | 20 | 13 | 33 |
| 2015 November | 28 | 19 | 47 |
| 2015 October | 34 | 20 | 54 |
| 2015 September | 33 | 14 | 47 |
| 2015 August | 39 | 6 | 45 |
| 2015 July | 41 | 8 | 49 |
| 2015 June | 25 | 1 | 26 |
| 2015 May | 36 | 7 | 43 |
| 2015 April | 45 | 14 | 59 |
| 2015 March | 44 | 11 | 55 |
| 2015 February | 28 | 4 | 32 |
| 2015 January | 23 | 5 | 28 |
| 2014 December | 19 | 10 | 29 |
| 2014 November | 27 | 8 | 35 |
| 2014 October | 23 | 10 | 33 |
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