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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histology revealed labial edema with slight ectasia of the lymphatic vessels in the specimen&#44; with no granulomatous lesions or other inflammatory lesions &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;10&#41;&#46;</p>"
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    "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&#44; including Melkersson-Rosenthal syndrome or its monosymptomatic form&#44; granulomatous cheilitis or Miescher syndrome&#46;</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&#46; Since its onset about 2 years previously&#44; the lesion had been completely stable and asymptomatic&#46; The patient&#39;s personal history included dyslipidemia&#44; treated with atorvastatin&#44; and left hemifacial spasm 8 years earlier&#46; She had also received regular 6-month infiltrations of botulinum toxin in the orbicular muscle of the left eyelid for 4 years&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The examination revealed well-organized&#44; nonpitting&#44; nonpulsatile edema in the lower left lip&#44; of soft&#44; rubbery consistency&#59; the mucous membranes and other oral structures were completely normal&#46; No discoloration or local signs of superinfection were observed &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Additionally&#44; the patient was unable to fully close her left eye and presented muscle twitching on the left hemiface&#46; She reported no surgical operations&#44; local radiotherapy&#44; infiltrations of botulinum toxin or filler material in the area&#44; or prosthetic dental work in contact with the affected area&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Because the lip swelling was accompanied by motor disturbances&#44; the lesion was biopsied to perform a differential diagnosis with granulomatous cheilitis&#46; Histology of the specimen showed lip edema with some ectasia of the lymphatic vessels in the specimen&#44; but no granulomatous lesions or other inflammatory lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</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&#46; The clinical entity is characterized by repeated tonic&#8211;clonic twitches of the muscles innervated by the facial nerve &#40;cranial nerve VII&#41;&#46; 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&#46; However&#44; cases secondary to large vascular abnormalities and tumors &#40;e&#46;g&#46;&#44; neurinoma or meningioma&#41; have also been reported&#46; The condition typically presents insidiously and affects the orbicular muscles&#44; in the form of eyelid twitching&#44; and extends ipsilaterally to other muscles&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Spasms are initially paroxysmal&#44; but later increase in frequency and severity and then become sustained contractions&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> These tonic contractions would&#44; in our opinion&#44; hinder venous and lymph return from the affected areas&#44; thus causing an increase in the diameter of the abundant lip vasculature&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The differential diagnosis should include conditions such as hereditary and acquired angioedema &#40;in these cases&#44; the lesions disappear within 24 to 48<span class="elsevierStyleHsp" style=""></span>hours&#41;&#44; various orofacial granulomatoses such as lymphomatoid granulomatosis&#44; Crohn disease&#44; and Melkerson-Rosenthal syndrome and its monosymptomatic form &#40;granulomatous cheilitis or Miescher syndrome&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> The contractions can be associated with other accompanying manifestations &#40;e&#46;g&#46;&#44; pulmonary or intestinal involvement&#44; facial paralysis&#44; or tongue fissures&#44; respectively&#41;&#44; and the biopsy tends to provide the diagnosis&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Likewise&#44; it is necessary to rule out congenital or acquired lip malformations &#40;lymphangioma&#44; hemangioma&#44; and neurofibromatosis&#41;&#44; Ascher syndrome &#40;possibly associated with blepharochalasis and thyroid goiter&#41;&#44; and cheilitis glandularis&#44; where the increased size of lips is due to secondary inflammation of heterotopic salivary glands in the lips&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> All these conditions have a well-established histologic diagnosis&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">A final hypothesis to consider is the effect of regular botulinum toxin infiltration&#46; Although cases of local edema secondary to botulinum toxin infiltrations have been described&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> in our patient this treatment was never administered in the circumoral region&#44; and edema severity was unchanged long after injections&#44; when the drug would have been expected to have a lower therapeutic effect&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In conclusion&#44; although hemifacial spasm is not rare&#44; there are no published reports in the literature that associate it with persistent labial edema&#46; This phenomenon could be explained by the fact that the condition is asymptomatic and mundane&#46; Therefore&#44; the patient is unlikely to be referred to a specialist unless the clinical picture is noteworthy&#44; hence the condition is underdiagnosed&#46; Because both manifestations can appear together&#44; they should be taken into account in the differential diagnosis of persistent lip swelling&#46;</p></span>"
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Case and Research Letters
Persistent Labial Edema Secondary to Hemifacial Spasm
Edema labial persistente secundario a espasmo hemifacial
R. Bella-Navarro
Corresponding author
rbellanavarro@gmail.com

Corresponding author.
, V. Alonso-Usero, E.M. Gutiérrez Paredes, E. Jordá-Cuevas
Servicio de Dermatología, Hospital Clínico Universitario, Valencia, Spain
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histology revealed labial edema with slight ectasia of the lymphatic vessels in the specimen&#44; with no granulomatous lesions or other inflammatory lesions &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;10&#41;&#46;</p>"
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    "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&#44; including Melkersson-Rosenthal syndrome or its monosymptomatic form&#44; granulomatous cheilitis or Miescher syndrome&#46;</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&#46; Since its onset about 2 years previously&#44; the lesion had been completely stable and asymptomatic&#46; The patient&#39;s personal history included dyslipidemia&#44; treated with atorvastatin&#44; and left hemifacial spasm 8 years earlier&#46; She had also received regular 6-month infiltrations of botulinum toxin in the orbicular muscle of the left eyelid for 4 years&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The examination revealed well-organized&#44; nonpitting&#44; nonpulsatile edema in the lower left lip&#44; of soft&#44; rubbery consistency&#59; the mucous membranes and other oral structures were completely normal&#46; No discoloration or local signs of superinfection were observed &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Additionally&#44; the patient was unable to fully close her left eye and presented muscle twitching on the left hemiface&#46; She reported no surgical operations&#44; local radiotherapy&#44; infiltrations of botulinum toxin or filler material in the area&#44; or prosthetic dental work in contact with the affected area&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Because the lip swelling was accompanied by motor disturbances&#44; the lesion was biopsied to perform a differential diagnosis with granulomatous cheilitis&#46; Histology of the specimen showed lip edema with some ectasia of the lymphatic vessels in the specimen&#44; but no granulomatous lesions or other inflammatory lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</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&#46; The clinical entity is characterized by repeated tonic&#8211;clonic twitches of the muscles innervated by the facial nerve &#40;cranial nerve VII&#41;&#46; 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&#46; However&#44; cases secondary to large vascular abnormalities and tumors &#40;e&#46;g&#46;&#44; neurinoma or meningioma&#41; have also been reported&#46; The condition typically presents insidiously and affects the orbicular muscles&#44; in the form of eyelid twitching&#44; and extends ipsilaterally to other muscles&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Spasms are initially paroxysmal&#44; but later increase in frequency and severity and then become sustained contractions&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> These tonic contractions would&#44; in our opinion&#44; hinder venous and lymph return from the affected areas&#44; thus causing an increase in the diameter of the abundant lip vasculature&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The differential diagnosis should include conditions such as hereditary and acquired angioedema &#40;in these cases&#44; the lesions disappear within 24 to 48<span class="elsevierStyleHsp" style=""></span>hours&#41;&#44; various orofacial granulomatoses such as lymphomatoid granulomatosis&#44; Crohn disease&#44; and Melkerson-Rosenthal syndrome and its monosymptomatic form &#40;granulomatous cheilitis or Miescher syndrome&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> The contractions can be associated with other accompanying manifestations &#40;e&#46;g&#46;&#44; pulmonary or intestinal involvement&#44; facial paralysis&#44; or tongue fissures&#44; respectively&#41;&#44; and the biopsy tends to provide the diagnosis&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Likewise&#44; it is necessary to rule out congenital or acquired lip malformations &#40;lymphangioma&#44; hemangioma&#44; and neurofibromatosis&#41;&#44; Ascher syndrome &#40;possibly associated with blepharochalasis and thyroid goiter&#41;&#44; and cheilitis glandularis&#44; where the increased size of lips is due to secondary inflammation of heterotopic salivary glands in the lips&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> All these conditions have a well-established histologic diagnosis&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">A final hypothesis to consider is the effect of regular botulinum toxin infiltration&#46; Although cases of local edema secondary to botulinum toxin infiltrations have been described&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> in our patient this treatment was never administered in the circumoral region&#44; and edema severity was unchanged long after injections&#44; when the drug would have been expected to have a lower therapeutic effect&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In conclusion&#44; although hemifacial spasm is not rare&#44; there are no published reports in the literature that associate it with persistent labial edema&#46; This phenomenon could be explained by the fact that the condition is asymptomatic and mundane&#46; Therefore&#44; the patient is unlikely to be referred to a specialist unless the clinical picture is noteworthy&#44; hence the condition is underdiagnosed&#46; Because both manifestations can appear together&#44; they should be taken into account in the differential diagnosis of persistent lip swelling&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara">Please cite this article as&#58; Bella-Navarro R&#44; et al&#46; Edema labial persistente secundario a espasmo hemifacial&#46; Actas Dermosifiliogr&#46; 2012&#59;103&#58;157&#8211;158&#46;</p>"
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ISSN: 15782190
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