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"apellidos" => "Beltrán Blasco" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0213485313001266" "doi" => "10.1016/j.nrl.2013.06.001" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0213485313001266?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173580815000218?idApp=UINPBA00004N" "url" => "/21735808/0000003000000003/v2_201503250259/S2173580815000218/v2_201503250259/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Harlequin syndrome, a rare neurological disease" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "185" "paginaFinal" => "187" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "R.J. Zabalza Estévez, F. Unanue López" "autores" => array:2 [ 0 => array:4 [ "nombre" => "R.J." "apellidos" => "Zabalza Estévez" "email" => array:1 [ 0 => "zabalzaes@telefonica.net" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "F." "apellidos" => "Unanue López" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Universitario Donostia, San Sebastián, Guipúzcoa, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Oftalmología, Hospital Universitario Donostia, San Sebastián, Guipúzcoa, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de Arlequín, una rareza neurológica" ] ] "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" => 1125 "Ancho" => 900 "Tamanyo" => 115984 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Left hemifacial flushing.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">In 1988, Lance et al.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">1</span></a> described a syndrome consisting of unilateral facial flushing and sweating triggered by exercise or hot weather. They called it ‘Harlequin syndrome’ (HS) based on Harlequin, a <span class="elsevierStyleItalic">Commedia dell’Arte</span> character. As a general rule, HS is a benign condition caused by a dysfunction in the upper thoracic sympathetic pathway. When it affects the first thoracic segment (T1), it is associated with ipsilateral Horner syndrome due to oculosympathetic fibre involvement. HS is a rare disease affecting autonomic innervation. Although benign in most cases, it is extremely distressing for patients and a diagnostic challenge for doctors.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a 54-year-old woman with long-standing right palpebral ptosis. She had no relevant personal or family history. Approximately 4 years earlier, she had noticed a narrowing of the right palpebral fissure, which was sometimes more pronounced with no apparent trigger factor. She had never experienced diplopia, dysphagia, excessive limb fatigue, or any other signs or symptoms suggesting a neuromuscular disease. The patient's medical records reported a 15-year history of left-sided facial flushing and profuse sweating with the right side of the face remaining pale and dry (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). These episodes were triggered by physical exercise and were especially noticeable in hotter settings. The patient described a distinct frontier between the two halves of her face, and regarded the prominent left-sided facial flushing as a source of social embarrassment. Physical examination revealed mild right palpebral ptosis and slight anisocoria with right miosis. Irises were the same colour, and pupillomotor response was normal. Results from the general and neurological examinations were normal. In light of the above, we suspected a lesion in the sympathetic pathway affecting vasomotor and sudomotor fibres in the right side of the face (consistent with HS), possibly associated with Horner syndrome. Results from the analyses (complete blood count, erythrocyte sedimentation rate, glucose test, a serum electrolyte study, renal and thyroid function studies, etc.) were normal. Neuroimaging studies (cranial and cervical MRI, duplex Doppler ultrasonography of the supra-aortic trunks, and thoracic computerized tomography) showed no relevant abnormalities. A negative apraclonidine test ruled out Horner syndrome. However, instillation of phenylephrine 10% reversed ptosis, and the patient was diagnosed with aponeurotic ptosis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Once we determined the patient showed no structural changes, she was diagnosed with idiopathic HS and informed that it was a benign condition requiring no treatment. The patient was also offered surgical treatment for the aponeurotic ptosis but she refused since the condition was not functionally debilitating.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">HS is a rare autonomic disorder whose main symptom is the appearance of hemifacial flushing and sweating attacks, generally triggered by hot weather or physical exertion. Flushing and sweating does not occur on one side due to ipsilateral dysfunction of the vasomotor and sudomotor sympathetic pathways.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">2</span></a> Researchers postulate that compensatory neuronal activity on the healthy side accentuates the skin symptoms typical of the disease. Dysfunction may be located at any of the three levels in the cervical sympathetic chain. The first neuron originates in the hypothalamus. It descends along the brainstem into the spinal cord, where it forms a synapse with the second (preganglionic) neuron of the intermediolateral column, at the level of segments C8-T2. The preganglionic neuron leaves the spinal cord, travels through the stellate ganglion, and ascends to the superior cervical ganglion through the paravertebral sympathetic chain, which is in contact with the apex of the lung. The second (preganglionic) and third (postganglionic) neurons form a synapse in the superior cervical ganglion. Two branches leave this ganglion. One runs along the internal carotid artery and includes the vasomotor and sudomotor fibres innervating the nose and medial frontal region and the sympathetic fibres which cause the iris to dilate; the second branch runs along the external carotid artery and includes vasomotor and sudomotor postganglionic fibres that innervate the rest of the face.</p><p id="par0020" class="elsevierStylePara elsevierViewall">HS is usually benign. It has been described in up to 10% of healthy newborns and it is associated with a temporary imbalance in the tone of cutaneous blood vessels due to hypothalamic immaturity.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> However, many different aetiologies for HS have been described, including dissection of the carotid artery,<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">4</span></a> toxic goitre,<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">5</span></a> tumours,<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">6–8</span></a> syringomyelia,<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">9</span></a> multiple sclerosis,<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">10</span></a> and iatrogenic effects of invasive procedures.<a class="elsevierStyleCrossRefs" href="#bib0135"><span class="elsevierStyleSup">11–14</span></a> Researchers have recently attempted to find a pathogenic connection between migraine and HS based on the autonomic dysfunction associated with migraine. However, this correlation is yet to be established.<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">15</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The term ‘Harlequin syndrome’ should be used exclusively for attacks of flushing and sweating which are not associated with any other neurological symptoms. When these symptoms appear in conjunction with other autonomic disorders, the term ‘Harlequin sign’ should be used.<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">16</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The diagnostic process for patients with HS should attempt to determine the organic cause. If symptoms are idiopathic, it is essential to assure patients that the disorder is benign and that follow-up examinations will not be necessary.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Although idiopathic cases require no treatment, contralateral sympathectomy can be performed when HS has a severe psychological impact on patients. However, this surgical procedure may lead to compensatory flushing and sweating in other locations.<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">16</span></a></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: Zabalza Estévez RJ, Unanue López F. Síndrome de Arlequín, una rareza neurológica. Neurología. 2015;30:185–187.</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" => 1125 "Ancho" => 900 "Tamanyo" => 115984 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Left hemifacial flushing.</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" => 508 "Ancho" => 1000 "Tamanyo" => 76317 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(a) Right palpebral ptosis. 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Year/Month | Html | Total | |
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2024 October | 206 | 22 | 228 |
2024 September | 394 | 29 | 423 |
2024 August | 383 | 28 | 411 |
2024 July | 440 | 26 | 466 |
2024 June | 358 | 21 | 379 |
2024 May | 392 | 13 | 405 |
2024 April | 360 | 16 | 376 |
2024 March | 446 | 17 | 463 |
2024 February | 649 | 14 | 663 |
2024 January | 487 | 11 | 498 |
2023 December | 374 | 22 | 396 |
2023 November | 480 | 16 | 496 |
2023 October | 570 | 23 | 593 |
2023 September | 403 | 17 | 420 |
2023 August | 567 | 21 | 588 |
2023 July | 739 | 24 | 763 |
2023 June | 558 | 22 | 580 |
2023 May | 687 | 20 | 707 |
2023 April | 609 | 17 | 626 |
2023 March | 418 | 13 | 431 |
2023 February | 374 | 13 | 387 |
2023 January | 304 | 25 | 329 |
2022 December | 258 | 6 | 264 |
2022 November | 288 | 21 | 309 |
2022 October | 400 | 26 | 426 |
2022 September | 407 | 13 | 420 |
2022 August | 461 | 21 | 482 |
2022 July | 369 | 16 | 385 |
2022 June | 313 | 17 | 330 |
2022 May | 269 | 21 | 290 |
2022 April | 293 | 20 | 313 |
2022 March | 335 | 18 | 353 |
2022 February | 267 | 17 | 284 |
2022 January | 301 | 21 | 322 |
2021 December | 232 | 16 | 248 |
2021 November | 232 | 15 | 247 |
2021 October | 334 | 18 | 352 |
2021 September | 405 | 15 | 420 |
2021 August | 351 | 9 | 360 |
2021 July | 393 | 28 | 421 |
2021 June | 279 | 15 | 294 |
2021 May | 311 | 15 | 326 |
2021 April | 651 | 49 | 700 |
2021 March | 391 | 13 | 404 |
2021 February | 216 | 16 | 232 |
2021 January | 286 | 19 | 305 |
2020 December | 294 | 30 | 324 |
2020 November | 355 | 25 | 380 |
2020 October | 210 | 21 | 231 |
2020 September | 392 | 27 | 419 |
2020 August | 445 | 14 | 459 |
2020 July | 467 | 15 | 482 |
2020 June | 367 | 19 | 386 |
2020 May | 397 | 13 | 410 |
2020 April | 406 | 12 | 418 |
2020 March | 420 | 20 | 440 |
2020 February | 411 | 8 | 419 |
2020 January | 396 | 13 | 409 |
2019 December | 282 | 10 | 292 |
2019 November | 400 | 7 | 407 |
2019 October | 310 | 18 | 328 |
2019 September | 332 | 15 | 347 |
2019 August | 261 | 15 | 276 |
2019 July | 330 | 34 | 364 |
2019 June | 359 | 45 | 404 |
2019 May | 438 | 77 | 515 |
2019 April | 338 | 47 | 385 |
2019 March | 190 | 17 | 207 |
2019 February | 160 | 21 | 181 |
2019 January | 182 | 14 | 196 |
2018 December | 180 | 12 | 192 |
2018 November | 239 | 16 | 255 |
2018 October | 279 | 10 | 289 |
2018 September | 107 | 15 | 122 |
2018 August | 39 | 22 | 61 |
2018 July | 63 | 11 | 74 |
2018 June | 69 | 4 | 73 |
2018 May | 41 | 10 | 51 |
2018 April | 26 | 13 | 39 |
2018 March | 38 | 5 | 43 |
2018 February | 11 | 1 | 12 |
2018 January | 17 | 4 | 21 |
2017 December | 16 | 5 | 21 |
2017 November | 17 | 7 | 24 |
2017 October | 43 | 9 | 52 |
2017 September | 25 | 21 | 46 |
2017 August | 37 | 11 | 48 |
2017 July | 52 | 14 | 66 |
2017 June | 109 | 31 | 140 |
2017 May | 46 | 16 | 62 |
2017 April | 42 | 11 | 53 |
2017 March | 30 | 41 | 71 |
2017 February | 34 | 5 | 39 |
2017 January | 28 | 5 | 33 |
2016 December | 36 | 20 | 56 |
2016 November | 57 | 16 | 73 |
2016 October | 55 | 15 | 70 |
2016 September | 62 | 18 | 80 |
2016 August | 35 | 19 | 54 |
2016 July | 48 | 5 | 53 |
2016 June | 64 | 28 | 92 |
2016 May | 51 | 32 | 83 |
2016 April | 33 | 27 | 60 |
2016 March | 62 | 31 | 93 |
2016 February | 46 | 27 | 73 |
2016 January | 51 | 26 | 77 |
2015 December | 58 | 26 | 84 |
2015 November | 57 | 27 | 84 |
2015 October | 89 | 19 | 108 |
2015 September | 48 | 18 | 66 |
2015 August | 106 | 8 | 114 |
2015 July | 1702 | 15 | 1717 |
2015 June | 39 | 11 | 50 |
2015 May | 37 | 18 | 55 |
2015 April | 74 | 27 | 101 |
2015 March | 17 | 10 | 27 |