Case and Research Letters
Persistent Labial Edema Secondary to Hemifacial Spasm
Edema labial persistente secundario a espasmo hemifacial
R. Bella-Navarro
, V. Alonso-Usero, E.M. Gutiérrez Paredes, E. Jordá-Cuevas
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Servicio de Dermatología, Hospital Clínico Universitario, Valencia, Spain
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Bella-Navarro, V. Alonso-Usero, E.M. Gutiérrez Paredes, E. Jordá-Cuevas" "autores" => array:4 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Bella-Navarro" "email" => array:1 [ 0 => "rbellanavarro@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "V." "apellidos" => "Alonso-Usero" ] 2 => array:2 [ "nombre" => "E.M." "apellidos" => "Gutiérrez Paredes" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "Jordá-Cuevas" ] ] "afiliaciones" => array:1 [ 0 => array:1 [ "entidad" => "Servicio de Dermatología, Hospital Clínico Universitario, Valencia, Spain" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Edema labial persistente secundario a espasmo hemifacial" ] ] "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" => 2002 "Ancho" => 1500 "Tamanyo" => 466406 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histology revealed labial edema with slight ectasia of the lymphatic vessels in the specimen, with no granulomatous lesions or other inflammatory lesions (hematoxylin–eosin, original magnification ×10).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Persistent labial edema is a nonspecific clinical manifestation that can occur in many different diseases, including Melkersson-Rosenthal syndrome or its monosymptomatic form, granulomatous cheilitis or Miescher syndrome.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe the case of a 76-year-old woman who was referred to our department for acquired edema in the lower left lip. Since its onset about 2 years previously, the lesion had been completely stable and asymptomatic. The patient's personal history included dyslipidemia, treated with atorvastatin, and left hemifacial spasm 8 years earlier. She had also received regular 6-month infiltrations of botulinum toxin in the orbicular muscle of the left eyelid for 4 years.</p><p id="par0015" class="elsevierStylePara elsevierViewall">The examination revealed well-organized, nonpitting, nonpulsatile edema in the lower left lip, of soft, rubbery consistency; the mucous membranes and other oral structures were completely normal. No discoloration or local signs of superinfection were observed (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Additionally, the patient was unable to fully close her left eye and presented muscle twitching on the left hemiface. She reported no surgical operations, local radiotherapy, infiltrations of botulinum toxin or filler material in the area, or prosthetic dental work in contact with the affected area.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Because the lip swelling was accompanied by motor disturbances, the lesion was biopsied to perform a differential diagnosis with granulomatous cheilitis. Histology of the specimen showed lip edema with some ectasia of the lymphatic vessels in the specimen, but no granulomatous lesions or other inflammatory lesions (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Hemifacial spasm is an involuntary abnormal movement of the face and is relatively common. The clinical entity is characterized by repeated tonic–clonic twitches of the muscles innervated by the facial nerve (cranial nerve VII). Hemifacial spasm is seen more often in women and in adulthood and usually appears as the sequela of <span class="elsevierStyleItalic">idiopathic</span> peripheral facial paralysis. However, cases secondary to large vascular abnormalities and tumors (e.g., neurinoma or meningioma) have also been reported. The condition typically presents insidiously and affects the orbicular muscles, in the form of eyelid twitching, and extends ipsilaterally to other muscles.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Spasms are initially paroxysmal, but later increase in frequency and severity and then become sustained contractions.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> These tonic contractions would, in our opinion, hinder venous and lymph return from the affected areas, thus causing an increase in the diameter of the abundant lip vasculature.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The differential diagnosis should include conditions such as hereditary and acquired angioedema (in these cases, the lesions disappear within 24 to 48<span class="elsevierStyleHsp" style=""></span>hours), various orofacial granulomatoses such as lymphomatoid granulomatosis, Crohn disease, and Melkerson-Rosenthal syndrome and its monosymptomatic form (granulomatous cheilitis or Miescher syndrome).<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3–5</span></a> The contractions can be associated with other accompanying manifestations (e.g., pulmonary or intestinal involvement, facial paralysis, or tongue fissures, respectively), and the biopsy tends to provide the diagnosis.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Likewise, it is necessary to rule out congenital or acquired lip malformations (lymphangioma, hemangioma, and neurofibromatosis), Ascher syndrome (possibly associated with blepharochalasis and thyroid goiter), and cheilitis glandularis, where the increased size of lips is due to secondary inflammation of heterotopic salivary glands in the lips.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> All these conditions have a well-established histologic diagnosis.</p><p id="par0040" class="elsevierStylePara elsevierViewall">A final hypothesis to consider is the effect of regular botulinum toxin infiltration. Although cases of local edema secondary to botulinum toxin infiltrations have been described,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> in our patient this treatment was never administered in the circumoral region, and edema severity was unchanged long after injections, when the drug would have been expected to have a lower therapeutic effect.</p><p id="par0045" class="elsevierStylePara elsevierViewall">In conclusion, although hemifacial spasm is not rare, there are no published reports in the literature that associate it with persistent labial edema. This phenomenon could be explained by the fact that the condition is asymptomatic and mundane. Therefore, the patient is unlikely to be referred to a specialist unless the clinical picture is noteworthy, hence the condition is underdiagnosed. Because both manifestations can appear together, they should be taken into account in the differential diagnosis of persistent lip swelling.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Bella-Navarro R, et al. Edema labial persistente secundario a espasmo hemifacial. Actas Dermosifiliogr. 2012;103:157–158.</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" => 1093 "Ancho" => 1587 "Tamanyo" => 156160 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Soft edema in the lower left lip.</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" => 2002 "Ancho" => 1500 "Tamanyo" => 466406 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histology revealed labial edema with slight ectasia of the lymphatic vessels in the specimen, with no granulomatous lesions or other inflammatory lesions (hematoxylin–eosin, original magnification ×10).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Hemifacial spasm: a review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "R.H. 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Terashima" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.jaad.2009.06.040" "Revista" => array:6 [ "tituloSerie" => "J Am Acad Dermatol" "fecha" => "2009" "volumen" => "61" "paginaInicial" => "961" "paginaFinal" => "970" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/19744746" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15782190/0000010300000002/v1_201304241253/S1578219012000686/v1_201304241253/en/main.assets" "Apartado" => array:4 [ "identificador" => "6169" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Cases and Research Letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15782190/0000010300000002/v1_201304241253/S1578219012000686/v1_201304241253/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219012000686?idApp=UINPBA000044" ]
| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 3 | 4 | 7 |
| 2024 Octubre | 99 | 53 | 152 |
| 2024 Septiembre | 138 | 40 | 178 |
| 2024 Agosto | 164 | 55 | 219 |
| 2024 Julio | 169 | 42 | 211 |
| 2024 Junio | 148 | 43 | 191 |
| 2024 Mayo | 124 | 39 | 163 |
| 2024 Abril | 146 | 28 | 174 |
| 2024 Marzo | 146 | 36 | 182 |
| 2024 Febrero | 144 | 30 | 174 |
| 2024 Enero | 127 | 49 | 176 |
| 2023 Diciembre | 149 | 24 | 173 |
| 2023 Noviembre | 182 | 31 | 213 |
| 2023 Octubre | 107 | 26 | 133 |
| 2023 Septiembre | 117 | 36 | 153 |
| 2023 Agosto | 84 | 30 | 114 |
| 2023 Julio | 77 | 55 | 132 |
| 2023 Junio | 105 | 35 | 140 |
| 2023 Mayo | 86 | 47 | 133 |
| 2023 Abril | 63 | 37 | 100 |
| 2023 Marzo | 113 | 41 | 154 |
| 2023 Febrero | 83 | 29 | 112 |
| 2023 Enero | 61 | 33 | 94 |
| 2022 Diciembre | 70 | 41 | 111 |
| 2022 Noviembre | 56 | 23 | 79 |
| 2022 Octubre | 42 | 25 | 67 |
| 2022 Septiembre | 30 | 25 | 55 |
| 2022 Agosto | 26 | 27 | 53 |
| 2022 Julio | 39 | 35 | 74 |
| 2022 Junio | 27 | 36 | 63 |
| 2022 Mayo | 79 | 34 | 113 |
| 2022 Abril | 61 | 34 | 95 |
| 2022 Marzo | 73 | 49 | 122 |
| 2022 Febrero | 51 | 36 | 87 |
| 2022 Enero | 55 | 44 | 99 |
| 2021 Diciembre | 43 | 37 | 80 |
| 2021 Noviembre | 73 | 45 | 118 |
| 2021 Octubre | 137 | 56 | 193 |
| 2021 Septiembre | 92 | 46 | 138 |
| 2021 Agosto | 72 | 34 | 106 |
| 2021 Julio | 48 | 33 | 81 |
| 2021 Junio | 61 | 39 | 100 |
| 2021 Mayo | 70 | 41 | 111 |
| 2021 Abril | 111 | 66 | 177 |
| 2021 Marzo | 99 | 33 | 132 |
| 2021 Febrero | 75 | 31 | 106 |
| 2021 Enero | 50 | 13 | 63 |
| 2020 Diciembre | 34 | 17 | 51 |
| 2020 Noviembre | 43 | 18 | 61 |
| 2020 Octubre | 23 | 7 | 30 |
| 2020 Septiembre | 30 | 11 | 41 |
| 2020 Agosto | 32 | 13 | 45 |
| 2020 Julio | 19 | 17 | 36 |
| 2020 Junio | 61 | 35 | 96 |
| 2020 Mayo | 26 | 25 | 51 |
| 2020 Abril | 37 | 23 | 60 |
| 2020 Marzo | 35 | 20 | 55 |
| 2020 Febrero | 2 | 9 | 11 |
| 2020 Enero | 1 | 11 | 12 |
| 2019 Diciembre | 4 | 3 | 7 |
| 2019 Octubre | 0 | 2 | 2 |
| 2019 Septiembre | 4 | 3 | 7 |
| 2019 Agosto | 0 | 10 | 10 |
| 2019 Julio | 0 | 12 | 12 |
| 2019 Junio | 2 | 17 | 19 |
| 2019 Mayo | 2 | 35 | 37 |
| 2019 Abril | 1 | 19 | 20 |
| 2019 Marzo | 2 | 6 | 8 |
| 2019 Febrero | 0 | 2 | 2 |
| 2019 Enero | 2 | 6 | 8 |
| 2018 Diciembre | 2 | 1 | 3 |
| 2018 Noviembre | 2 | 0 | 2 |
| 2018 Octubre | 2 | 0 | 2 |
| 2018 Septiembre | 1 | 0 | 1 |
| 2018 Agosto | 0 | 7 | 7 |
| 2018 Julio | 0 | 8 | 8 |
| 2018 Junio | 0 | 2 | 2 |
| 2018 Mayo | 0 | 7 | 7 |
| 2018 Abril | 0 | 9 | 9 |
| 2018 Marzo | 2 | 0 | 2 |
| 2018 Febrero | 53 | 6 | 59 |
| 2018 Enero | 66 | 11 | 77 |
| 2017 Diciembre | 72 | 8 | 80 |
| 2017 Noviembre | 54 | 6 | 60 |
| 2017 Octubre | 65 | 5 | 70 |
| 2017 Septiembre | 65 | 2 | 67 |
| 2017 Agosto | 126 | 7 | 133 |
| 2017 Julio | 98 | 9 | 107 |
| 2017 Junio | 118 | 9 | 127 |
| 2017 Mayo | 108 | 7 | 115 |
| 2017 Abril | 92 | 7 | 99 |
| 2017 Marzo | 61 | 7 | 68 |
| 2017 Febrero | 47 | 9 | 56 |
| 2017 Enero | 44 | 12 | 56 |
| 2016 Diciembre | 69 | 16 | 85 |
| 2016 Noviembre | 120 | 18 | 138 |
| 2016 Octubre | 98 | 21 | 119 |
| 2016 Septiembre | 111 | 10 | 121 |
| 2016 Agosto | 95 | 13 | 108 |
| 2016 Julio | 68 | 9 | 77 |
| 2016 Junio | 5 | 8 | 13 |
| 2016 Mayo | 12 | 9 | 21 |
| 2016 Abril | 14 | 19 | 33 |
| 2016 Marzo | 9 | 7 | 16 |
| 2016 Febrero | 5 | 10 | 15 |
| 2016 Enero | 12 | 17 | 29 |
| 2015 Diciembre | 10 | 5 | 15 |
| 2015 Noviembre | 16 | 10 | 26 |
| 2015 Octubre | 12 | 7 | 19 |
| 2015 Septiembre | 9 | 0 | 9 |
| 2015 Agosto | 19 | 1 | 20 |
| 2015 Julio | 76 | 12 | 88 |
| 2015 Junio | 48 | 6 | 54 |
| 2015 Mayo | 105 | 13 | 118 |
| 2015 Abril | 107 | 4 | 111 |
| 2015 Marzo | 75 | 10 | 85 |
| 2015 Febrero | 44 | 11 | 55 |
| 2015 Enero | 43 | 14 | 57 |
| 2014 Diciembre | 44 | 9 | 53 |
| 2014 Noviembre | 36 | 15 | 51 |
| 2014 Octubre | 54 | 15 | 69 |
| 2014 Septiembre | 44 | 10 | 54 |
| 2014 Agosto | 69 | 16 | 85 |
| 2014 Julio | 60 | 18 | 78 |
| 2014 Junio | 81 | 14 | 95 |
| 2014 Mayo | 78 | 20 | 98 |
| 2014 Abril | 63 | 10 | 73 |
| 2014 Marzo | 73 | 16 | 89 |
| 2014 Febrero | 18 | 8 | 26 |
| 2014 Enero | 23 | 11 | 34 |
| 2013 Diciembre | 20 | 6 | 26 |
| 2013 Noviembre | 16 | 12 | 28 |
| 2013 Octubre | 15 | 3 | 18 |
| 2013 Septiembre | 13 | 9 | 22 |
| 2013 Agosto | 18 | 9 | 27 |
| 2013 Julio | 11 | 7 | 18 |
| 2013 Junio | 11 | 13 | 24 |
| 2013 Mayo | 14 | 14 | 28 |
| 2013 Abril | 18 | 18 | 36 |
| 2013 Marzo | 17 | 8 | 25 |
| 2013 Febrero | 39 | 8 | 47 |
| 2013 Enero | 45 | 4 | 49 |
| 2012 Diciembre | 28 | 4 | 32 |
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