Case and research letters
Pemphigoid Gestationis: Therapeutic Response to Pre- and Postpartum Immunoglobulin Therapy
Penfigoide gestacional. Respuesta terapéutica a inmunoglobulinas pre y postparto
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Respuesta terapéutica a inmunoglobulinas pre y postparto" "tienePdf" => "es" "tieneTextoCompleto" => "es" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "735" "paginaFinal" => "737" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Pemphigoid Gestationis: Therapeutic Response to Pre- and Postpartum Immunoglobulin Therapy" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 969 "Ancho" => 1396 "Tamanyo" => 121849 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A. Erupción papulosa con ampollas a tensión en zona abdominal de nuestra paciente. B. 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Ruiz-Villaverde, D. Sánchez-Cano, C.L. Ramirez-Tortosa" "autores" => array:3 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Ruiz-Villaverde" "email" => array:1 [ 0 => "ismenios@hotmail.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" => "D." "apellidos" => "Sánchez-Cano" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "C.L." "apellidos" => "Ramirez-Tortosa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Unidad de Dermatología, Complejo Hospitalario Ciudad de Jaén, Jaén, Spain" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "UGC Anatomía Patológica, Complejo Hospitalario Ciudad de Jaén, Jaén, Spain" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Penfigoide gestacional. Respuesta terapéutica a inmunoglobulinas pre y postparto" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 970 "Ancho" => 1397 "Tamanyo" => 121273 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Papular eruption with tense blisters on the patient's abdomen. B, Condition of the patient at 6 weeks after delivery.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pemphigoid gestationis or herpes gestationis is a subepidermal blistering disease that occurs in women in the second or third trimesters of pregnancy or even during puerperium. It is a rare skin disease whose incidence has been estimated at around 1 case in every 40 000 to 60 000 pregnancies. It is more common in patients with HLA DR3 and DR4 haplotypes.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Although most cases respond well to oral corticosteroids, some can be resistant to this and other treatments. We report a case in which we successfully applied intravenous immunoglobulin treatment.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 25-year-old, primiparous woman with hypertension on treatment with enalapril, presented in the second trimester of pregnancy, at 22 weeks, with a rash of intensely pruritic plaques that started in the periumbilical region and subsequently appeared on the anterior aspect of both wrists and extensor surfaces of the lower limbs. Laboratory and serological tests (hepatitis B and C viruses, cytomegalovirus, and Epstein-Barr virus) showed no abnormalities except for mild eosinophilia. Tense blisters developed on several of the lesions (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). Histology revealed edema with an infiltrate formed of lymphocytes and abundant eosinophils (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A), and direct immunofluorescence showed linear deposits of C3 at the dermoepidermal junction (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B). Pemphigoid gestationis was diagnosed and treatment was initiated with prednisone (30<span class="elsevierStyleHsp" style=""></span>mg/d). The dose was increased to 60<span class="elsevierStyleHsp" style=""></span>mg/day and subsequently to 75<span class="elsevierStyleHsp" style=""></span>mg/d due to the lack of response on weekly follow-up.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In the final month of pregnancy intravenous immunoglobulin was administered at a dose of 500 mg/kg/d for 5 days. We succeeded in reducing the dose of prednisone to 30<span class="elsevierStyleHsp" style=""></span>mg/kg/d (reducing 5<span class="elsevierStyleHsp" style=""></span>mg/wk until that dose was reached) with no worsening of symptoms.</p><p id="par0020" class="elsevierStylePara elsevierViewall">At 15 days after delivery, the dose of prednisone was dropped to 15<span class="elsevierStyleHsp" style=""></span>mg/d. In agreement with the gynecology department of our hospital, a new course of intravenous immunoglobulin was initiated at the same dose in order to prevent a probable resurgence of the condition, given the poor response. At 18 weeks after delivery, prednisone was suspended with no recurrence of lesions in the following 6 months (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Follow-up laboratory tests and blood pressure values showed no variation either during or after treatment.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The treatment of choice in pemphigoid gestationis is oral corticosteroids. The usual dose is around 0.5<span class="elsevierStyleHsp" style=""></span>mg/kg/d of prednisone by mouth, which rapidly improves symptoms in most cases. However, systemic corticosteroid therapy is sometimes insufficient to control the disorder, as occurred in our patient. Other treatments have been tested, including plasmapheresis, intravenous immunoglobulin, pyridoxine, ritodrine, and immunosuppressant drugs such as cyclosporin,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> azathioprine, dapsone, and tacrolimus.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The use of intravenous immunoglobulin in autoimmune bullous diseases resistant to conventional treatment is increasing, since it offers a convenient alternative treatment with few side effects, most of which are mild and self-limiting. Episodes of flushing, myalgia, headaches, nausea and vomiting, tachycardia, and a slight increase in blood pressure have been reported in most cases between 30 and 60<span class="elsevierStyleHsp" style=""></span>minutes after starting infusion.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Anaphylaxis is very rare and has mainly occurred in patients with immunoglobulin A deficiency. These adverse effects are almost always avoidable with premedication according to an established protocol at the medical day care units where the infusion is administered.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The literature reports few cases of the use of intravenous immunoglobulin in pemphigoid gestationis,<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5–8</span></a> though it has been employed successfully to manage other blistering diseases such as pemphigus vulgaris, bullous pemphigoid, and acquired epidermolysis bullosa.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> The mechanism of action of intravenous immunoglobulin is not fully understood but it appears to act at different points in the immune cascade, including functional blockade of Fc receptors on splenic macrophages, inhibition of complement-mediated actions, and modulation of cytokine production.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In the absence of large patient series, given the low incidence of this pathology, the dose and time of administration of immunoglobulin vary according to the author reviewed. No significant differences were detected between regimens of 3 or 5 days, and most authors followed existing protocols at their hospitals. We considered applying this treatment pre- and postpartum in order to control skin disease in the final stages of pregnancy and prevent a possible recurrence during the puerperium.Jolles et al<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> observed that treatment with intravenous immunoglobulin in autoimmune bullous diseases is always more successful when used as adjuvant therapy (91%) rather than monotherapy (51%). Subsequently published cases of pemphigoid gestationis report therapeutic success in both situations.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5–8</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">In our patient, we used intravenous immunoglobulin associated with systemic corticosteroids, owing to the difficulty of controlling the skin symptoms and taking into account that corticosteroids during pregnancy can involve risk for the fetus (growth retardation, prematurity) and for the mother (osteonecrosis, hypertension, infections, etc.). We believe that the regimen followed in our patient may be of interest in cases with an indolent clinical course and that are refractory to conventional treatment.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Ruiz-Villaverde R, et al. Penfigoide gestacional. Respuesta terapéutica a inmunoglobulinas pre y postparto. Actas Dermosifiliogr. 2011;102:735-747.</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" => 970 "Ancho" => 1397 "Tamanyo" => 121273 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Papular eruption with tense blisters on the patient's abdomen. B, Condition of the patient at 6 weeks after delivery.</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" => 1026 "Ancho" => 1393 "Tamanyo" => 261252 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Subepidermal blister with eosinophils (hematoxylin-eosin, original magnification x10). B, Direct immunofluorescence: linear deposits of C3.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clinical experience in pemphigoid gestationis: report of 10 cases" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "L.A. Castro" 1 => "R.B. Lundell" 2 => "P.K. Krause" 3 => "L.E. 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Pratt" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "J Cutan Med Surg" "fecha" => "2010" "volumen" => "14" "paginaInicial" => "189" "paginaFinal" => "192" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20642990" "web" => "Medline" ] ] ] ] ] ] ] ] 5 => array:3 [ "identificador" => "bib0030" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Persistent herpes gestationis treated with high-dose intravenous immunoglobulin" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "S. Rodrigues Cdos" 1 => "P. Filipe" 2 => "M. Solana Mdel" 3 => "L. Soares de Almeida" 4 => "J. Cirne de Castro" 5 => "M.M. 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Black" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Clin Exp Dermatol" "fecha" => "1998" "volumen" => "23" "paginaInicial" => "185" "paginaFinal" => "188" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/9894365" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15782190/0000010200000009/v1_201304241238/S1578219011000916/v1_201304241238/en/main.assets" "Apartado" => array:4 [ "identificador" => "6157" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case and Research Letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15782190/0000010200000009/v1_201304241238/S1578219011000916/v1_201304241238/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219011000916?idApp=UINPBA000044" ]
| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 14 | 11 | 25 |
| 2024 Octubre | 114 | 45 | 159 |
| 2024 Septiembre | 112 | 23 | 135 |
| 2024 Agosto | 132 | 61 | 193 |
| 2024 Julio | 120 | 41 | 161 |
| 2024 Junio | 101 | 36 | 137 |
| 2024 Mayo | 86 | 41 | 127 |
| 2024 Abril | 91 | 25 | 116 |
| 2024 Marzo | 83 | 47 | 130 |
| 2024 Febrero | 66 | 43 | 109 |
| 2024 Enero | 87 | 48 | 135 |
| 2023 Diciembre | 97 | 16 | 113 |
| 2023 Noviembre | 98 | 28 | 126 |
| 2023 Octubre | 103 | 25 | 128 |
| 2023 Septiembre | 104 | 31 | 135 |
| 2023 Agosto | 87 | 18 | 105 |
| 2023 Julio | 114 | 40 | 154 |
| 2023 Junio | 109 | 21 | 130 |
| 2023 Mayo | 153 | 37 | 190 |
| 2023 Abril | 138 | 27 | 165 |
| 2023 Marzo | 133 | 21 | 154 |
| 2023 Febrero | 170 | 19 | 189 |
| 2023 Enero | 119 | 44 | 163 |
| 2022 Diciembre | 112 | 52 | 164 |
| 2022 Noviembre | 37 | 21 | 58 |
| 2022 Octubre | 30 | 17 | 47 |
| 2022 Septiembre | 44 | 43 | 87 |
| 2022 Agosto | 58 | 32 | 90 |
| 2022 Julio | 33 | 32 | 65 |
| 2022 Junio | 40 | 24 | 64 |
| 2022 Mayo | 114 | 36 | 150 |
| 2022 Abril | 106 | 41 | 147 |
| 2022 Marzo | 96 | 57 | 153 |
| 2022 Febrero | 106 | 29 | 135 |
| 2022 Enero | 192 | 44 | 236 |
| 2021 Diciembre | 87 | 50 | 137 |
| 2021 Noviembre | 87 | 39 | 126 |
| 2021 Octubre | 89 | 46 | 135 |
| 2021 Septiembre | 66 | 37 | 103 |
| 2021 Agosto | 94 | 29 | 123 |
| 2021 Julio | 103 | 29 | 132 |
| 2021 Junio | 82 | 26 | 108 |
| 2021 Mayo | 68 | 57 | 125 |
| 2021 Abril | 121 | 78 | 199 |
| 2021 Marzo | 104 | 25 | 129 |
| 2021 Febrero | 69 | 28 | 97 |
| 2021 Enero | 65 | 26 | 91 |
| 2020 Diciembre | 70 | 18 | 88 |
| 2020 Noviembre | 46 | 13 | 59 |
| 2020 Octubre | 30 | 11 | 41 |
| 2020 Septiembre | 31 | 17 | 48 |
| 2020 Agosto | 30 | 20 | 50 |
| 2020 Julio | 34 | 18 | 52 |
| 2020 Junio | 50 | 27 | 77 |
| 2020 Mayo | 27 | 15 | 42 |
| 2020 Abril | 40 | 31 | 71 |
| 2020 Marzo | 39 | 34 | 73 |
| 2020 Febrero | 4 | 11 | 15 |
| 2020 Enero | 0 | 5 | 5 |
| 2019 Diciembre | 4 | 4 | 8 |
| 2019 Noviembre | 0 | 1 | 1 |
| 2019 Septiembre | 4 | 7 | 11 |
| 2019 Agosto | 0 | 3 | 3 |
| 2019 Julio | 0 | 7 | 7 |
| 2019 Junio | 2 | 8 | 10 |
| 2019 Mayo | 0 | 10 | 10 |
| 2019 Abril | 0 | 7 | 7 |
| 2019 Marzo | 2 | 8 | 10 |
| 2019 Febrero | 0 | 3 | 3 |
| 2019 Enero | 3 | 0 | 3 |
| 2018 Noviembre | 1 | 0 | 1 |
| 2018 Octubre | 3 | 0 | 3 |
| 2018 Septiembre | 1 | 0 | 1 |
| 2018 Junio | 0 | 1 | 1 |
| 2018 Mayo | 0 | 6 | 6 |
| 2018 Abril | 0 | 1 | 1 |
| 2018 Marzo | 14 | 2 | 16 |
| 2018 Febrero | 76 | 5 | 81 |
| 2018 Enero | 130 | 8 | 138 |
| 2017 Diciembre | 153 | 10 | 163 |
| 2017 Noviembre | 85 | 10 | 95 |
| 2017 Octubre | 49 | 9 | 58 |
| 2017 Septiembre | 69 | 9 | 78 |
| 2017 Agosto | 91 | 12 | 103 |
| 2017 Julio | 76 | 6 | 82 |
| 2017 Junio | 99 | 15 | 114 |
| 2017 Mayo | 99 | 9 | 108 |
| 2017 Abril | 80 | 16 | 96 |
| 2017 Marzo | 78 | 37 | 115 |
| 2017 Febrero | 61 | 15 | 76 |
| 2017 Enero | 47 | 9 | 56 |
| 2016 Diciembre | 88 | 14 | 102 |
| 2016 Noviembre | 128 | 10 | 138 |
| 2016 Octubre | 127 | 18 | 145 |
| 2016 Septiembre | 188 | 18 | 206 |
| 2016 Agosto | 140 | 15 | 155 |
| 2016 Julio | 61 | 15 | 76 |
| 2016 Junio | 8 | 16 | 24 |
| 2016 Mayo | 8 | 15 | 23 |
| 2016 Abril | 9 | 7 | 16 |
| 2016 Marzo | 10 | 8 | 18 |
| 2016 Febrero | 12 | 16 | 28 |
| 2016 Enero | 8 | 12 | 20 |
| 2015 Diciembre | 15 | 5 | 20 |
| 2015 Noviembre | 24 | 1 | 25 |
| 2015 Octubre | 14 | 3 | 17 |
| 2015 Septiembre | 7 | 10 | 17 |
| 2015 Agosto | 8 | 4 | 12 |
| 2015 Julio | 66 | 10 | 76 |
| 2015 Junio | 54 | 10 | 64 |
| 2015 Mayo | 85 | 15 | 100 |
| 2015 Abril | 61 | 10 | 71 |
| 2015 Marzo | 46 | 14 | 60 |
| 2015 Febrero | 47 | 11 | 58 |
| 2015 Enero | 45 | 15 | 60 |
| 2014 Diciembre | 45 | 8 | 53 |
| 2014 Noviembre | 31 | 6 | 37 |
| 2014 Octubre | 52 | 20 | 72 |
| 2014 Septiembre | 63 | 12 | 75 |
| 2014 Agosto | 50 | 19 | 69 |
| 2014 Julio | 70 | 20 | 90 |
| 2014 Junio | 99 | 14 | 113 |
| 2014 Mayo | 104 | 19 | 123 |
| 2014 Abril | 77 | 10 | 87 |
| 2014 Marzo | 85 | 21 | 106 |
| 2014 Febrero | 54 | 8 | 62 |
| 2014 Enero | 52 | 22 | 74 |
| 2013 Diciembre | 67 | 11 | 78 |
| 2013 Noviembre | 39 | 18 | 57 |
| 2013 Octubre | 10 | 6 | 16 |
| 2013 Septiembre | 11 | 11 | 22 |
| 2013 Agosto | 20 | 11 | 31 |
| 2013 Julio | 11 | 10 | 21 |
| 2013 Junio | 10 | 13 | 23 |
| 2013 Mayo | 11 | 6 | 17 |
| 2013 Abril | 21 | 14 | 35 |
| 2013 Marzo | 17 | 8 | 25 |
| 2013 Febrero | 33 | 2 | 35 |
| 2013 Enero | 20 | 6 | 26 |
| 2012 Diciembre | 16 | 5 | 21 |
| 2012 Noviembre | 0 | 1 | 1 |
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