Letter to the Editor
Unilateral eyelid myokymia as a form of presentation of multiple sclerosis
Mioquimias palpebrales unilaterales como forma de presentación de una esclerosis múltiple
a Servicio de Neurología, Hospital Universitario Valle de Hebrón, Universidad Autónoma de Barcelona, Barcelona, Spain
b Área Salut Básica Sant Josep, ICS, L’Hospitalet de Llobregat, Barcelona, Spain
c Servicio de Neurología, IDIBELL Hospital Universitario de Bellvitge, L’Hospitalet de Llobregat, Barcelona, Spain
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Palasí, N. Martínez-Sánchez, L. Bau, J. Campdelacreu" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Palasí" "email" => array:1 [ 0 => "toni.palasi@gmail.com" ] "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" => "N." "apellidos" => "Martínez-Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "L." "apellidos" => "Bau" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Campdelacreu" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Universitario Valle de Hebrón, Universidad Autónoma de Barcelona, Barcelona, Spain" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Área Salut Básica Sant Josep, ICS, L’Hospitalet de Llobregat, Barcelona, Spain" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Neurología, IDIBELL Hospital Universitario de Bellvitge, L’Hospitalet de Llobregat, Barcelona, Spain" "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Mioquimias palpebrales unilaterales como forma de presentación de una esclerosis múltiple" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 625 "Ancho" => 1799 "Tamanyo" => 125711 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cranial MRI was repeated at 4 months. Axial FLAIR sequences (a, b) and a T1-weighted axial sequence with gadolinium contrast (c) showed new lesions that were not apparent in the previous study, including lesions with gadolinium ring enhancement (arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Myokymias are localised involuntary contractions that are wavelike or vermicular and propagate through affected striated muscle.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> They are caused by simultaneous or sequential activations of 1, 2, or more motor units of a muscle. An electromyography reading shows spontaneous muscle activity with different motor units producing brief, repetitive discharges of action potentials in rhythmic or semi-rhythmic bursts. This is followed by a short (0.5 to 3<span class="elsevierStyleHsp" style=""></span>seconds) and frequently irregular interval of electric silence before the following myokymic discharge occurs.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">When myokymia occurs in the face, the most frequently affected muscle is the orbicularis oculi. Myokymia in this muscle results in small, visible contractions of part of the eyelid, typically the lower eyelid. Eyelid myokymias (EM) tend to be transient and self-limiting within a few days of onset. They present in young, healthy subjects with no associated diseases.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,6,7</span></a> We describe a patient in whom the presence of continuous EM resulted in a diagnosis of multiple sclerosis (MS).</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was a 34-year-old woman with no relevant medical history or drug use. She was examined for abnormal movements compatible with myokymias in the right lower eyelid which had been occurring for 2 months. The symptoms initially presented sporadically during the day in episodes lasting from a few minutes to an hour. As the weeks passed, their frequency and intensity had increased and muscle twitches had become continuous. The first neurological examination revealed generalised hyperreflexia; all other findings were normal.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Given the intensity of the myokymias, the patient began treatment with carbamazepine (800<span class="elsevierStyleHsp" style=""></span>mg/day), but the drug had to be discontinued quickly due to the appearance of a maculopapular rash and elevated transaminases.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The symptoms resolved spontaneously 9 weeks after their onset. A week after EMs had become continuous, doctors performed a brain MRI which showed multiple lesions in supratentorial white matter in both hemispheres, semioval centres, and the juxtaventricular zone, plus a lesion in the posterior right frontal lobe (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A complete blood study including serology and autoimmune markers yielded normal results. CSF analysis indicated the presence of oligoclonal bands, although they were not found in serum. A second cranial MRI taken at 4 months revealed new lesions, including several with gadolinium uptake (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Based on these findings, the patient was diagnosed with MS.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Facial myokymias that occur continuously and affect all the muscles on one side of the face have often been described in association with different types of lesions of the ipsilateral pontine tegmentum,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–6,9</span></a> especially tumours (gliomas or metastasis),<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,5</span></a> cysticercosis,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> and MS. In MS, myokymias may occur either throughout the duration of the disease<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,9,10</span></a> or appear as its first symptom.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Other entities with which EM is less frequently associated are subarachnoid haemorrhage,<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> multiple system atrophy, and Guillain-Barré syndrome.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3,5</span></a> In MS, facial myokymias tend to be self-limiting in the course of a few weeks (typically between 2 weeks and 6 months)<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,10,12</span></a> and they rarely last as long as a year.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> However, myokymias secondary to malignant tumours typically last several years and resolve with treatment of the neoplasia.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,5,10</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Unlike facial myokymias, those limited to the eyelid only tend to appear in healthy young subjects. They are associated with stress, fatigue, exercise, and excessive caffeine use.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,7,12</span></a> On rare occasions, they may be associated with underlying disease or extend to other facial muscles. In a typical presentation, they are unilateral and transient, appearing in episodes lasting a few minutes at a time over the course of a few days or weeks. It is very uncommon for EMs to appear continuously during periods spanning months, and they would still be considered benign even in such cases.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,7</span></a> However, available evidence is scarce. There is only 1 study evaluating persistent long-term EM,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> and this study found that only 1 of its study population of 15 developed clinically defined MS. It concluded that associations between long-term EM and other neurological diseases are uncommon. As we were unable to identify any of the trigger factors described for EM in our patient, and the condition persisted, we performed 2 cranial MRI scans and a lumber puncture which resulted in a diagnosis of MS.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Cranial MRI study of our patient was unable to show any lesions specific to the ipsilateral pons. Some cases of hemifacial myokymias are described in which cranial MRI scans did not show typical lesions in the pons.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,6,9</span></a> One possible reason is that the lesion causing facial myokymia in MS tends to resolve when clinical symptoms have stopped appearing.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> This was observed in a study that used cranial MRI to re-examine patients in whom typical lesions had previously been detected and whose symptoms had resolved. In 6 of the 8 patients, the lesions had disappeared within periods ranging from 1 week to 44 months.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> A cranial MRI was performed on our patient a week after her referral to our unit. Another possible mechanism could be hyperexcitability of the intra-axial part of facial nerve fibres due to possible functional deafferentation of inhibitory pathways of the facial nerve nucleus caused by demyelinating lesions on the supranuclear level.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,9,13</span></a> As a result, myokymias would not necessarily have to be caused by lesions in the pons.</p><p id="par0045" class="elsevierStylePara elsevierViewall">In conclusion, we feel that while EM is a benign entity, it may be indicative of an underlying neurological disease, such as MS, if it persists over an extended period of time. We recommend conservative treatment for these patients at first, and elimination of known triggers such as caffeine, tobacco, and alcohol. If the EM continues, imaging studies should be completed in order to rule out any underlying lesions.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Palasí A, et al. Mioquimias palpebrales unilaterales como forma de presentación de una esclerosis múltiple. Neurología. 2013;28:187–9.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara">Part of this study was presented in poster format at the 15th annual meeting of the Catalan Society of Neurology.</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" => 741 "Ancho" => 1401 "Tamanyo" => 124298 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cranial MRI. T2-weighted axial sequences (a) and sagittal FLAIR sequences (b) reveal multiple hyperintense lesions in supratentorial white matter.</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" => 625 "Ancho" => 1799 "Tamanyo" => 125711 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cranial MRI was repeated at 4 months. Axial FLAIR sequences (a, b) and a T1-weighted axial sequence with gadolinium contrast (c) showed new lesions that were not apparent in the previous study, including lesions with gadolinium ring enhancement (arrow).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:13 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Persistent facial myokymia: a rare pathognomic physical sign of intrinsic brain-stem lesions: report of 2 cases and review of literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "R.R. Sharma" 1 => "N.V. Mathad" 2 => "D.N. Joshi" 3 => "T.B. Mazarelo" 4 => "M.M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 59 | 0 | 59 |
| 2024 October | 497 | 14 | 511 |
| 2024 September | 678 | 27 | 705 |
| 2024 August | 682 | 20 | 702 |
| 2024 July | 800 | 14 | 814 |
| 2024 June | 545 | 6 | 551 |
| 2024 May | 615 | 5 | 620 |
| 2024 April | 665 | 19 | 684 |
| 2024 March | 840 | 19 | 859 |
| 2024 February | 856 | 12 | 868 |
| 2024 January | 962 | 13 | 975 |
| 2023 December | 827 | 9 | 836 |
| 2023 November | 836 | 11 | 847 |
| 2023 October | 923 | 25 | 948 |
| 2023 September | 698 | 4 | 702 |
| 2023 August | 642 | 6 | 648 |
| 2023 July | 702 | 9 | 711 |
| 2023 June | 740 | 12 | 752 |
| 2023 May | 987 | 14 | 1001 |
| 2023 April | 880 | 11 | 891 |
| 2023 March | 844 | 9 | 853 |
| 2023 February | 577 | 8 | 585 |
| 2023 January | 599 | 15 | 614 |
| 2022 December | 527 | 24 | 551 |
| 2022 November | 655 | 18 | 673 |
| 2022 October | 618 | 18 | 636 |
| 2022 September | 612 | 21 | 633 |
| 2022 August | 615 | 31 | 646 |
| 2022 July | 501 | 22 | 523 |
| 2022 June | 482 | 28 | 510 |
| 2022 May | 470 | 19 | 489 |
| 2022 April | 585 | 16 | 601 |
| 2022 March | 637 | 26 | 663 |
| 2022 February | 617 | 26 | 643 |
| 2022 January | 626 | 26 | 652 |
| 2021 December | 548 | 22 | 570 |
| 2021 November | 549 | 28 | 577 |
| 2021 October | 612 | 43 | 655 |
| 2021 September | 558 | 31 | 589 |
| 2021 August | 649 | 31 | 680 |
| 2021 July | 775 | 20 | 795 |
| 2021 June | 663 | 21 | 684 |
| 2021 May | 770 | 18 | 788 |
| 2021 April | 1674 | 24 | 1698 |
| 2021 March | 1001 | 21 | 1022 |
| 2021 February | 766 | 34 | 800 |
| 2021 January | 672 | 34 | 706 |
| 2020 December | 667 | 41 | 708 |
| 2020 November | 730 | 35 | 765 |
| 2020 October | 529 | 17 | 546 |
| 2020 September | 1082 | 19 | 1101 |
| 2020 August | 894 | 20 | 914 |
| 2020 July | 747 | 18 | 765 |
| 2020 June | 957 | 17 | 974 |
| 2020 May | 1107 | 31 | 1138 |
| 2020 April | 952 | 14 | 966 |
| 2020 March | 943 | 10 | 953 |
| 2020 February | 1074 | 20 | 1094 |
| 2020 January | 1036 | 19 | 1055 |
| 2019 December | 804 | 14 | 818 |
| 2019 November | 872 | 13 | 885 |
| 2019 October | 804 | 20 | 824 |
| 2019 September | 839 | 13 | 852 |
| 2019 August | 709 | 20 | 729 |
| 2019 July | 1034 | 16 | 1050 |
| 2019 June | 726 | 38 | 764 |
| 2019 May | 769 | 50 | 819 |
| 2019 April | 842 | 15 | 857 |
| 2019 March | 496 | 8 | 504 |
| 2019 February | 521 | 15 | 536 |
| 2019 January | 601 | 14 | 615 |
| 2018 December | 410 | 10 | 420 |
| 2018 November | 273 | 9 | 282 |
| 2018 October | 223 | 14 | 237 |
| 2018 September | 262 | 27 | 289 |
| 2018 August | 113 | 1 | 114 |
| 2018 July | 87 | 4 | 91 |
| 2018 June | 84 | 3 | 87 |
| 2018 May | 124 | 3 | 127 |
| 2018 April | 131 | 2 | 133 |
| 2018 March | 180 | 2 | 182 |
| 2018 February | 92 | 3 | 95 |
| 2018 January | 92 | 3 | 95 |
| 2017 December | 89 | 0 | 89 |
| 2017 November | 112 | 4 | 116 |
| 2017 October | 123 | 2 | 125 |
| 2017 September | 101 | 9 | 110 |
| 2017 August | 108 | 3 | 111 |
| 2017 July | 138 | 1 | 139 |
| 2017 June | 153 | 15 | 168 |
| 2017 May | 139 | 9 | 148 |
| 2017 April | 170 | 12 | 182 |
| 2017 March | 117 | 35 | 152 |
| 2017 February | 246 | 2 | 248 |
| 2017 January | 117 | 5 | 122 |
| 2016 December | 121 | 10 | 131 |
| 2016 November | 200 | 11 | 211 |
| 2016 October | 248 | 7 | 255 |
| 2016 September | 263 | 10 | 273 |
| 2016 August | 179 | 7 | 186 |
| 2016 July | 86 | 2 | 88 |
| 2016 June | 127 | 11 | 138 |
| 2016 May | 119 | 27 | 146 |
| 2016 April | 110 | 31 | 141 |
| 2016 March | 124 | 29 | 153 |
| 2016 February | 117 | 11 | 128 |
| 2016 January | 93 | 7 | 100 |
| 2015 December | 90 | 6 | 96 |
| 2015 November | 93 | 12 | 105 |
| 2015 October | 105 | 7 | 112 |
| 2015 September | 87 | 5 | 92 |
| 2015 August | 94 | 5 | 99 |
| 2015 July | 102 | 10 | 112 |
| 2015 June | 69 | 4 | 73 |
| 2015 May | 85 | 4 | 89 |
| 2015 April | 75 | 6 | 81 |
| 2015 March | 72 | 9 | 81 |
| 2015 February | 20 | 4 | 24 |
| 2015 January | 28 | 9 | 37 |
| 2014 December | 37 | 9 | 46 |
| 2014 November | 25 | 5 | 30 |
| 2014 October | 28 | 6 | 34 |
| 2014 September | 26 | 3 | 29 |
| 2014 August | 46 | 4 | 50 |
| 2014 July | 29 | 3 | 32 |
| 2014 June | 30 | 2 | 32 |
| 2014 May | 33 | 3 | 36 |
| 2014 April | 20 | 3 | 23 |
| 2014 March | 20 | 3 | 23 |
| 2014 February | 34 | 6 | 40 |
| 2014 January | 28 | 4 | 32 |
| 2013 December | 27 | 7 | 34 |
| 2013 November | 56 | 5 | 61 |
| 2013 October | 55 | 2 | 57 |
| 2013 September | 50 | 1 | 51 |
| 2013 August | 64 | 5 | 69 |
| 2013 July | 36 | 6 | 42 |
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