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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cheilitis granulomatosa or Miescher syndrome &#40;MS&#41; is an uncommon disorder of unknown etiology and pathogenesis&#44; characterized by recurrent swelling of the lips&#46; It is included in the spectrum of orofacial granulomatosis&#44; the paradigm of which is Melkersson&#8211;Rosenthal syndrome&#44; in the cases in which it is accompanied by involvement of the facial nerve together with fissured tongue&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 45-year-old woman with a history of hypertension who had been treated for several years with atenolol&#46; She presented complaining of recurrent episodes of labial edema accompanied by burning pain&#46; The patient explained that she felt generally unwell&#44; in the absence of fever or chills&#46; She did not have constitutional symptoms&#44; diarrhea or abdominal pain&#44; and denied exposure to toxic agents and was unable to provide epidemiological data of interest&#46; She had not begun to take any new drugs&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Physical examination revealed the presence of lip swelling&#44; in the absence of ulcers or other associated lesions&#46; It also found that her facial nerve and tongue morphology were absolutely normal&#46; The results of cardiopulmonary evaluation were also normal&#46; A biopsy of the upper lip showed the presence of granulomatous dermatitis with a mixed component of predominantly superficial and deep perivascular infiltrate&#46; In the biopsy&#44; the findings with stains for acid&#8211;alcohol-resistant bacilli and fungi were negative&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">We performed the intradermal Mantoux test and the result was negative&#46; The findings were the same for the syphilis test &#40;Venereal Disease Research Laboratory &#91;VDRL&#93;&#41; and for serological tests for hepatitis B and C viruses&#44; and for human immunodeficiency virus&#46; The systemic study was accompanied by analyses to determine angiotensin-converting enzyme&#44; antinuclear antibodies&#44; nucleolar antibodies&#44; total extractable nuclear antibodies&#44; antineutrophil cytoplasmic antibodies&#44; immunoglobulins and erythrocyte sedimentation rate&#44; in which all of the results were normal or negative&#46; A chest radiograph showed a normal mediastinal silhouette&#44; and the absence of lymphadenopathy&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The response to glucocorticoids was very favorable&#44; and resulted in the disappearance of swelling and episodes of labial edema&#46; However&#44; 6 weeks later&#44; the patient came back with similar clinical signs&#44; that coincided with the decrease in steroid dose&#46; Treatment was begun with dapsone&#44; followed by an improvement in the symptoms&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Miescher syndrome appears to be an incomplete form of Melkersson&#8211;Rosenthal syndrome in 28&#37; of the patients&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> In general&#44; MS is an uncommon disease with special predilection for the second or third decades of life&#44; although cases have been reported at any age&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> It characteristically affects the upper lip and has a lesser effect on the lower lip&#44; although there have been anecdotal reports of the involvement of other regions of the oral cavity&#46; The most frequent presentation is acute and symptomatic swelling of the lips that lasts for hours or days&#46; The confirmation of the diagnosis is the discovery of non-necrotizing granulomas and perivascular lymphocyte infiltration in the biopsy of the affected region&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Other diseases with a granulomatous etiology&#44; such as sarcoidosis&#44; tuberculosis and inflammatory bowel disease&#44; should be taken into account in the differential diagnosis and must be ruled out&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">There is no ideal treatment for this disease&#46; A number of therapeutic strategies have been proposed&#44; including intralesional or systemic glucocorticoids&#44; as well as other options such as oral tetracyclines&#44; thalidomide&#44; dapsone or biological therapy &#40;anti-tumor necrosis factor <span class="elsevierStyleItalic">&#945;</span>&#41;&#44; with differing results&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Although it is rare&#44; MS should be taken into account in the differential diagnosis of recurrent edema of the lips&#46; A histological study is essential for the definitive diagnosis of the disease&#46; Treatment with corticosteroids is highly effective&#46; However&#44; as occurs with other immune-mediated diseases&#44; treatment of the underlying disease should be included&#46; Despite the few cases in which the use of dapsone is reported&#44; this drug at a dose of 50<span class="elsevierStyleHsp" style=""></span>mg a day&#44; may prove to be a useful alternative in MS&#44; as is illustrated by the experience we report here&#46;</p></span>"
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Letter to the Editor
Miescher Syndrome: An Uncommon Cause of Recurrent Swelling of the Lips
Síndrome de Miescher: una causa poco frecuente de tumefacción labial
Andrés González-Garcíaa,
Corresponding author
, Ignacio Barbolla Díaza, Walter Alberto Sifuentes Giraldob, José Luis Patier-de la Peñaa
a Servicio de Medicina Interna, Hospital Universitario Ramón y Cajal, Madrid, Spain
b Servicio de Reumatología, Hospital Universitario Ramón y Cajal, Madrid, Spain
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        "titulo" => "S&#237;ndrome de Miescher&#58; una causa poco frecuente de tumefacci&#243;n labial"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cheilitis granulomatosa or Miescher syndrome &#40;MS&#41; is an uncommon disorder of unknown etiology and pathogenesis&#44; characterized by recurrent swelling of the lips&#46; It is included in the spectrum of orofacial granulomatosis&#44; the paradigm of which is Melkersson&#8211;Rosenthal syndrome&#44; in the cases in which it is accompanied by involvement of the facial nerve together with fissured tongue&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 45-year-old woman with a history of hypertension who had been treated for several years with atenolol&#46; She presented complaining of recurrent episodes of labial edema accompanied by burning pain&#46; The patient explained that she felt generally unwell&#44; in the absence of fever or chills&#46; She did not have constitutional symptoms&#44; diarrhea or abdominal pain&#44; and denied exposure to toxic agents and was unable to provide epidemiological data of interest&#46; She had not begun to take any new drugs&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Physical examination revealed the presence of lip swelling&#44; in the absence of ulcers or other associated lesions&#46; It also found that her facial nerve and tongue morphology were absolutely normal&#46; The results of cardiopulmonary evaluation were also normal&#46; A biopsy of the upper lip showed the presence of granulomatous dermatitis with a mixed component of predominantly superficial and deep perivascular infiltrate&#46; In the biopsy&#44; the findings with stains for acid&#8211;alcohol-resistant bacilli and fungi were negative&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">We performed the intradermal Mantoux test and the result was negative&#46; The findings were the same for the syphilis test &#40;Venereal Disease Research Laboratory &#91;VDRL&#93;&#41; and for serological tests for hepatitis B and C viruses&#44; and for human immunodeficiency virus&#46; The systemic study was accompanied by analyses to determine angiotensin-converting enzyme&#44; antinuclear antibodies&#44; nucleolar antibodies&#44; total extractable nuclear antibodies&#44; antineutrophil cytoplasmic antibodies&#44; immunoglobulins and erythrocyte sedimentation rate&#44; in which all of the results were normal or negative&#46; A chest radiograph showed a normal mediastinal silhouette&#44; and the absence of lymphadenopathy&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The response to glucocorticoids was very favorable&#44; and resulted in the disappearance of swelling and episodes of labial edema&#46; However&#44; 6 weeks later&#44; the patient came back with similar clinical signs&#44; that coincided with the decrease in steroid dose&#46; Treatment was begun with dapsone&#44; followed by an improvement in the symptoms&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Miescher syndrome appears to be an incomplete form of Melkersson&#8211;Rosenthal syndrome in 28&#37; of the patients&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> In general&#44; MS is an uncommon disease with special predilection for the second or third decades of life&#44; although cases have been reported at any age&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> It characteristically affects the upper lip and has a lesser effect on the lower lip&#44; although there have been anecdotal reports of the involvement of other regions of the oral cavity&#46; The most frequent presentation is acute and symptomatic swelling of the lips that lasts for hours or days&#46; The confirmation of the diagnosis is the discovery of non-necrotizing granulomas and perivascular lymphocyte infiltration in the biopsy of the affected region&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Other diseases with a granulomatous etiology&#44; such as sarcoidosis&#44; tuberculosis and inflammatory bowel disease&#44; should be taken into account in the differential diagnosis and must be ruled out&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">There is no ideal treatment for this disease&#46; A number of therapeutic strategies have been proposed&#44; including intralesional or systemic glucocorticoids&#44; as well as other options such as oral tetracyclines&#44; thalidomide&#44; dapsone or biological therapy &#40;anti-tumor necrosis factor <span class="elsevierStyleItalic">&#945;</span>&#41;&#44; with differing results&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Although it is rare&#44; MS should be taken into account in the differential diagnosis of recurrent edema of the lips&#46; A histological study is essential for the definitive diagnosis of the disease&#46; Treatment with corticosteroids is highly effective&#46; However&#44; as occurs with other immune-mediated diseases&#44; treatment of the underlying disease should be included&#46; Despite the few cases in which the use of dapsone is reported&#44; this drug at a dose of 50<span class="elsevierStyleHsp" style=""></span>mg a day&#44; may prove to be a useful alternative in MS&#44; as is illustrated by the experience we report here&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Gonz&#225;lez-Garc&#237;a A&#44; Barbolla D&#237;az I&#44; Sifuentes Giraldo WA&#44; Patier-de la Pe&#241;a JL&#46; S&#237;ndrome de Miescher&#58; una causa poco frecuente de tumefacci&#243;n labial&#46; Reumatol Clin&#46; 2017&#59;13&#58;363&#8211;364&#46;</p>"
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