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1</a>&#41;&#46; Skin biopsy revealed a subepidermal blister with eosinophilic spongiosis&#44; without vacuolar degeneration of the epidermal basement membrane &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Direct immunofluorescence showed linear focal deposits of complement component 3 &#40;C3&#41; in the basement membrane&#46; No anti-BP180 antibodies were detected by indirect immunofluorescence&#46; Serology for <span class="elsevierStyleItalic">Mycoplasma pneumoniae</span> was negative for immunoglobulin &#40;Ig&#41; M and IgG&#46; The patient began corticosteroid treatment with oral prednisone &#40;0&#46;75&#160;mg&#47;kg&#47;d&#41; and topical 0&#46;05&#37; clobetasol propionate cream&#46; The lesions progressively improved and finally disappeared 2&#160;months after the patient gave birth&#44; leaving residual pigmentation&#46; The baby was delivered without complications &#40;eg&#44; preterm birth&#44; low birth weight&#41; and without skin lesions&#46; After 6&#160;months of follow-up the patient remained free of lesions&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Pemphigoid gestationis&#44; formerly known as herpes gestationis&#44; is a rare autoimmune blistering disease associated with pregnancy&#46; It usually occurs during the second or third trimester of pregnancy &#40;68&#37; of cases&#41;&#44; with incidence ranging from 1 per 20&#44;000 to 1 per 50&#44;000 population&#44; and tends to resolve after childbirth&#44; although flare-ups can occur after delivery and during menstruation&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The lesions begin as pruritic urticarial papules and plaques that rapidly evolve to vesicles but rarely progress to large blisters&#46; In up to 65&#37; of cases lesions initially appear in the periumbilical area&#44; subsequently spreading to other areas including the abdomen&#44; legs&#44; arms&#44; palms&#44; and soles&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> A case series by Hallaji and coworkers<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> reported no instances of mucosal involvement&#46; In another case series&#44; Castro et al<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> reported involvement of the hard palate mucosa in 1 patient and of the genital mucosa in another&#46; However&#44; neither of those articles specify whether the aforementioned involvement consisted of erosions&#44; ulcers&#44; blisters&#44; or vesicles&#46; Another peculiarity in our case was the appearance on the patient&#39;s palms and soles of exudative erythema multiforme-like lesions&#44; which are sporadically but infrequently described in the literature&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Histological findings depend on the intensity and stage of evolution of the lesions&#46; The presence of abundant eosinophils is a consistent finding in pemphigoid gestationis&#46; The key finding in the blistering phase is the presence of a subepidermal blister in the basement membrane&#46; In 100&#37; of cases direct immunofluorescence reveals the presence of linear C3 deposition along the basement membrane&#46; Deposition of IgG1 is observed in 25&#37; to 30&#37; of cases&#46; Depending on the technique used&#44; indirect immunofluorescence can detect IgG against the BP180 antigen in 30&#37; to 100&#37; of cases&#46; However&#44; titers do not always correlate with disease activity&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The first disease that should be included in the differential diagnosis is pruritic urticarial papules and plaques of pregnancy &#40;PUPPP&#41;&#46; The absence of striae gravidarum and the presence of periumbilical lesions in pemphigoid gestationis can facilitate distinction of pemphigoid gestationis from PUPPP&#46; Pemphigoid gestationis should also be distinguished from other blistering diseases that can occur during pregnancy&#44; including bullous pemphigoid&#44; dermatitis herpetiformis&#44; linear IgA disease&#44; bullous systemic lupus erythematosus&#44; exudative erythema multiforme&#44; toxic drug reaction&#44; and contact dermatitis&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Prednisolone &#40;0&#46;5&#8211;1&#160;mg&#47;kg&#47;d&#41; is one of the most effective and safest forms of treatment during pregnancy&#46; Prolonged systemic corticosteroid therapy can result in an increased incidence of preterm birth or low birth weight&#46; In cases of refractory and progressive pemphigoid gestationis other therapeutic options such as immunoglobulin or ciclosporin treatment can be considered&#44; always bearing in mind the potential risk to the fetus&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">4&#44;5</span></a> It should be noted that more severe flare-ups can occur during subsequent pregnancies or in conjunction with contraceptive use&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> In general the fetal prognosis is good&#58; mild involvement in the form of wheals or vesicles is reported in 10&#37; of cases&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion&#44; we present a case of pemphigoid gestationis&#44; confirmed by histology and direct immunofluorescence&#44; with extensive exudative erythema multiforme-like lesions and involvement of the oral mucosa&#46; Oral involvement in this type of blistering disease is very infrequent&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case and Research Letters
Pemphigoid Gestationis Mimicking Erythema Multiforme With Mucosal Involvement
Penfigoide gestacional simulando un eritema multiforme con afectación mucosa
A. Lobato-Berezoa,
Corresponding author
allobe@hotmail.es

Corresponding author.
, M.T. Fernández Figuerasc, J.A. Moreno Romerob, R.M. Pujola
a Departamento de Dermatología, Hospital del Mar-Parc Salut Mar, Barcelona, España
b Departamento de Dermatología, Hospital Universitari General de Catalunya, Sant Cugat del Vallès, Barcelona, España
c Departamento de Anatomía Patológica, Hospital Universitari General de Catalunya, Sant Cugat del Vallès, Barcelona, España
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pemphigoid gestationis is a relatively rare autoimmune blistering disease that occurs during pregnancy&#46; In very rare cases this process can be complicated by the appearance of generalized lesions with involvement of the oral mucosa&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A healthy&#44; 33-year-old woman in week 35 of her second pregnancy with the same partner was seen for generalized and buccal lesions&#46; The patient reported no previous infectious processes and had not begun treatment with any new medications&#46; Physical examination revealed large&#44; erythematous-edematous plaques around the umbilicus and on the neck&#46; Exudative erythema multiforme-like lesions were observed on the palms and soles&#44; and extensive erosions without vesicles or blisters on the oral mucosa &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Skin biopsy revealed a subepidermal blister with eosinophilic spongiosis&#44; without vacuolar degeneration of the epidermal basement membrane &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Direct immunofluorescence showed linear focal deposits of complement component 3 &#40;C3&#41; in the basement membrane&#46; No anti-BP180 antibodies were detected by indirect immunofluorescence&#46; Serology for <span class="elsevierStyleItalic">Mycoplasma pneumoniae</span> was negative for immunoglobulin &#40;Ig&#41; M and IgG&#46; The patient began corticosteroid treatment with oral prednisone &#40;0&#46;75&#160;mg&#47;kg&#47;d&#41; and topical 0&#46;05&#37; clobetasol propionate cream&#46; The lesions progressively improved and finally disappeared 2&#160;months after the patient gave birth&#44; leaving residual pigmentation&#46; The baby was delivered without complications &#40;eg&#44; preterm birth&#44; low birth weight&#41; and without skin lesions&#46; After 6&#160;months of follow-up the patient remained free of lesions&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Pemphigoid gestationis&#44; formerly known as herpes gestationis&#44; is a rare autoimmune blistering disease associated with pregnancy&#46; It usually occurs during the second or third trimester of pregnancy &#40;68&#37; of cases&#41;&#44; with incidence ranging from 1 per 20&#44;000 to 1 per 50&#44;000 population&#44; and tends to resolve after childbirth&#44; although flare-ups can occur after delivery and during menstruation&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The lesions begin as pruritic urticarial papules and plaques that rapidly evolve to vesicles but rarely progress to large blisters&#46; In up to 65&#37; of cases lesions initially appear in the periumbilical area&#44; subsequently spreading to other areas including the abdomen&#44; legs&#44; arms&#44; palms&#44; and soles&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> A case series by Hallaji and coworkers<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> reported no instances of mucosal involvement&#46; In another case series&#44; Castro et al<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> reported involvement of the hard palate mucosa in 1 patient and of the genital mucosa in another&#46; However&#44; neither of those articles specify whether the aforementioned involvement consisted of erosions&#44; ulcers&#44; blisters&#44; or vesicles&#46; Another peculiarity in our case was the appearance on the patient&#39;s palms and soles of exudative erythema multiforme-like lesions&#44; which are sporadically but infrequently described in the literature&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Histological findings depend on the intensity and stage of evolution of the lesions&#46; The presence of abundant eosinophils is a consistent finding in pemphigoid gestationis&#46; The key finding in the blistering phase is the presence of a subepidermal blister in the basement membrane&#46; In 100&#37; of cases direct immunofluorescence reveals the presence of linear C3 deposition along the basement membrane&#46; Deposition of IgG1 is observed in 25&#37; to 30&#37; of cases&#46; Depending on the technique used&#44; indirect immunofluorescence can detect IgG against the BP180 antigen in 30&#37; to 100&#37; of cases&#46; However&#44; titers do not always correlate with disease activity&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The first disease that should be included in the differential diagnosis is pruritic urticarial papules and plaques of pregnancy &#40;PUPPP&#41;&#46; The absence of striae gravidarum and the presence of periumbilical lesions in pemphigoid gestationis can facilitate distinction of pemphigoid gestationis from PUPPP&#46; Pemphigoid gestationis should also be distinguished from other blistering diseases that can occur during pregnancy&#44; including bullous pemphigoid&#44; dermatitis herpetiformis&#44; linear IgA disease&#44; bullous systemic lupus erythematosus&#44; exudative erythema multiforme&#44; toxic drug reaction&#44; and contact dermatitis&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Prednisolone &#40;0&#46;5&#8211;1&#160;mg&#47;kg&#47;d&#41; is one of the most effective and safest forms of treatment during pregnancy&#46; Prolonged systemic corticosteroid therapy can result in an increased incidence of preterm birth or low birth weight&#46; In cases of refractory and progressive pemphigoid gestationis other therapeutic options such as immunoglobulin or ciclosporin treatment can be considered&#44; always bearing in mind the potential risk to the fetus&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">4&#44;5</span></a> It should be noted that more severe flare-ups can occur during subsequent pregnancies or in conjunction with contraceptive use&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> In general the fetal prognosis is good&#58; mild involvement in the form of wheals or vesicles is reported in 10&#37; of cases&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">In conclusion&#44; we present a case of pemphigoid gestationis&#44; confirmed by histology and direct immunofluorescence&#44; with extensive exudative erythema multiforme-like lesions and involvement of the oral mucosa&#46; Oral involvement in this type of blistering disease is very infrequent&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Lobato-Berezo A&#44; Fern&#225;ndez Figueras MT&#44; Moreno Romero JA&#44; Pujol RM&#46; Pemphigoid Gestationis Mimicking Erythema Multiforme With Mucosal Involvement&#46; Actas Dermosifiliogr&#46; 2019&#59;110&#58;696&#8211;697&#46;</p>"
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Article information
ISSN: 15782190
Original language: English
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Idiomas
Actas Dermo-Sifiliográficas
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