Case and Research Letters
Narrowband UV-B Phototherapy in the Treatment of Generalized Hailey-Hailey Disease
Tratamiento de la enfermedad de Hailey-Hailey generalizada con fototerapia UVB de banda estrecha
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Álvarez-Chinchilla, I. Poveda Montoyo, B. Encabo-Durán, J. Bañuls Roca" "autores" => array:4 [ 0 => array:2 [ "nombre" => "P.J." "apellidos" => "Álvarez-Chinchilla" ] 1 => array:2 [ "nombre" => "I." "apellidos" => "Poveda Montoyo" ] 2 => array:2 [ "nombre" => "B." "apellidos" => "Encabo-Durán" ] 3 => array:2 [ "nombre" => "J." 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Complete resolution of the skin lesions after 60 sessions of UVB-NB therapy.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "D.A. De Luca, E.A. Zambrano, R.L. Galimberti, P.A. Enz" "autores" => array:4 [ 0 => array:2 [ "nombre" => "D.A." "apellidos" => "De Luca" ] 1 => array:2 [ "nombre" => "E.A." "apellidos" => "Zambrano" ] 2 => array:2 [ "nombre" => "R.L." "apellidos" => "Galimberti" ] 3 => array:2 [ "nombre" => "P.A." 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Abaca, L. Flores, V. Parra" "autores" => array:3 [ 0 => array:4 [ "nombre" => "M.C." "apellidos" => "Abaca" "email" => array:1 [ 0 => "celesteabacapdelallana@gmail.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" => "L." "apellidos" => "Flores" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "V." "apellidos" => "Parra" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Luis Lagomaggiore, Mendoza, Argentina" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Facultad de Ciencias Médicas, Universidad Nacional de Cuyo, Mendoza, Argentina" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tratamiento de la enfermedad de Hailey-Hailey generalizada con fototerapia UVB de banda estrecha" ] ] "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" => 703 "Ancho" => 1250 "Tamanyo" => 179458 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Macerated erythematous plaques.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Hailey-Hailey disease (HHD) is an autosomal dominant genodermatosis caused by a mutation in the <span class="elsevierStyleItalic">ATP2C1</span> gene. This gene encodes the hsPCA1 protein, which is responsible for calcium homeostasis. The mutation affects desmosome function, resulting in suprabasal acantholysis.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1–3</span></a> Clinically, it presents with recurrent flares of erythematous and macerated plaques in intertriginous areas, sometimes accompanied by erosions, fissures, and vesicles.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">4</span></a> Lesion superinfection and a foul odor, which can compromise patient quality of life, are observed in some cases.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">3</span></a> Generalized forms are infrequent and can be triggered by infections or drugs.<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">5,6</span></a> There are multiple available therapeutic options of variable efficacy.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1,7</span></a> Here, we describe complete lesion disappearance following narrowband ultraviolet B (NBUVB) phototherapy in a patient with generalized HHD resistant to multiple therapeutic regimens.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 41-year-old man with skin phototype <span class="elsevierStyleSmallCaps">III</span> and a history of histologically confirmed HHD that began at age 28 with involvement of the neck, axillae, and groin.</p><p id="par0015" class="elsevierStylePara elsevierViewall">He was seen for eroded and macerated erythematous plaques located in the skin folds. Over the preceding months the lesions, which caused mild pain and produced a foul odor, had spread to the trunk, upper extremities, and proximal thighs (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The lesions spread progressively despite treatment with 1% isoconazole and 2% mupirocin cream, 100<span class="elsevierStyleHsp" style=""></span>mg minocycline every 12<span class="elsevierStyleHsp" style=""></span>hours, and 150<span class="elsevierStyleHsp" style=""></span>mg fluconazole per day for 7 days, in repeated cycles.</p><p id="par0025" class="elsevierStylePara elsevierViewall">In the absence of a clinical response 3 weeks after starting treatment with prednisone (40<span class="elsevierStyleHsp" style=""></span>mg/d), NBUVB phototherapy was added to the regimen. NBUVB irradiation was carried out using a Waldmann F85/100W-01 cabinet equipped with TL01 lamps. The patient underwent a total of 8 weekly sessions, starting at a dose of 300<span class="elsevierStyleHsp" style=""></span>mJ/cm<span class="elsevierStyleSup">2</span> and increasing by 50<span class="elsevierStyleHsp" style=""></span>mJ/cm<span class="elsevierStyleSup">2</span> per week. The maximum dose of 500<span class="elsevierStyleHsp" style=""></span>mJ/cm<span class="elsevierStyleSup">2</span> was reached after the fifth session, and was maintained until the eighth week. The patient received a cumulative dose of 3500<span class="elsevierStyleHsp" style=""></span>mJ/cm<span class="elsevierStyleSup">2</span>. During each session he was positioned standing with the lower limbs in abduction and the upper limbs elevated above the level of the shoulders, thus allowing irradiation of the skin folds. No adverse effects were observed during treatment.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Four weeks after starting phototherapy a marked improvement was observed; the lesion extension decreased and the odor subsided. Consequently, the prednisone dose was progressively decreased at a rate of 5<span class="elsevierStyleHsp" style=""></span>mg per week, and prednisone treatment was ultimately discontinued 1 month after cessation of phototherapy.</p><p id="par0035" class="elsevierStylePara elsevierViewall">After completing the 8 phototherapy sessions the lesions resolved completely, leaving only residual hyperpigmentation (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">Currently, the patient is being treated with 1% clotrimazole and 0.1% betamethasone cream once per day, and has not relapsed over 12 months of follow-up.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Conventional treatment of HHD consists of topical and oral corticosteroids, antibiotics and antifungals, and topical calcineurin inhibitors, combined with general measures.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> Typically, the therapeutic response is both temporary and partial. The use of methotrexate, cyclosporine, thalidomide, retinoids, dapsone, botulinum toxin, dermabrasion, CO<span class="elsevierStyleInf">2</span> laser, alexandrite, erbium-doped yttrium aluminum garnet laser, radiotherapy, ultraviolet B (UV-B) phototherapy, and surgical treatment has been described,<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1,3,8</span></a> and photodynamic therapy has been proposed for the treatment of refractory cases.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">9</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In the case reported here, the patient presented with generalized HHD that responded poorly to corticosteroids in monotherapy, but responded well when NBUVB phototherapy was added to the treatment regimen. An improvement was observed 1 month after starting phototherapy and the lesions disappeared after 8 sessions over 2 months.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The use of NBUVB phototherapy for the treatment of HHD was first described in 1999 in a woman with psoriasis and concomitant HHD who was treated twice per week for 2 months with a suberythematogenic dose, to which she responded well.<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">10</span></a> Mizuno and coworkers<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">7</span></a> described the case of a woman with disseminated HHD who, after multiple treatment regimens without adequate response, underwent 8 sessions of NBUVB phototherapy using a similar regimen to that of our patient, and showed lesion improvement 2 weeks after completing treatment. The patient relapsed after treatment discontinuation, and once again showed a good response following its reintroduction. By contrast, our patient, who was treated for generalized HHD, showed no signs of recurrence up to 1 year after discontinuation of phototherapy. The lack of relapse over a long follow-up period may be because oral corticosteroid treatment had been initiated before NBUVB phototherapy began, and was continued, albeit at a lower dose, after completing the phototherapy regimen.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Classically, UV light is considered an aggravating factor of HDD because it stimulates production of proinflammatory cytokines, suppresses <span class="elsevierStyleItalic">ATP2C1</span> mRNA expression, and promotes acantholysis. Conversely, UV-B phototherapy decreases the number of epidermal T lymphocytes and dendritic cells, thereby attenuating cutaneous inflammation. In our patient, and in other cases described in the literature, phototherapy resulted in clinical improvement, suggesting the involvement of other factors in cases in which lesions worsen.<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">7,11</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">The present case is of particular interest because the patient in question had generalized HHD that responded poorly to conventional treatments but showed a good therapeutic response to NBUVB phototherapy. Based on these findings, we propose NBUVB phototherapy as a useful therapeutic alternative in patients with these characteristics.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of Interest" ] 1 => array:2 [ "identificador" => "xack382648" "titulo" => "Acknowledgments" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Abaca MC. Tratamiento de la enfermedad de Hailey-Hailey generalizada con fototerapia UVB de banda estrecha. Actas Dermosifiliogr. 2018;109:924–927.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 703 "Ancho" => 1250 "Tamanyo" => 179458 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Macerated erythematous plaques.</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" => 805 "Ancho" => 1300 "Tamanyo" => 151122 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Involvement of the chest, face, and upper arms.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 961 "Ancho" => 1300 "Tamanyo" => 225756 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Image showing lesion resolution in the axillary area after treatment with narrowband ultraviolet B (NBUVB) phototherapy.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:11 [ 0 => array:3 [ "identificador" => "bib0060" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Management of familial benign chronic pemphigus" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "H. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 9 | 8 | 17 |
| 2024 Octubre | 60 | 62 | 122 |
| 2024 Septiembre | 74 | 47 | 121 |
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| 2023 Noviembre | 83 | 24 | 107 |
| 2023 Octubre | 63 | 31 | 94 |
| 2023 Septiembre | 50 | 33 | 83 |
| 2023 Agosto | 46 | 24 | 70 |
| 2023 Julio | 61 | 27 | 88 |
| 2023 Junio | 40 | 24 | 64 |
| 2023 Mayo | 59 | 28 | 87 |
| 2023 Abril | 34 | 17 | 51 |
| 2023 Marzo | 52 | 32 | 84 |
| 2023 Febrero | 50 | 24 | 74 |
| 2023 Enero | 39 | 33 | 72 |
| 2022 Diciembre | 73 | 35 | 108 |
| 2022 Noviembre | 40 | 24 | 64 |
| 2022 Octubre | 94 | 32 | 126 |
| 2022 Septiembre | 22 | 42 | 64 |
| 2022 Agosto | 47 | 27 | 74 |
| 2022 Julio | 24 | 41 | 65 |
| 2022 Junio | 20 | 31 | 51 |
| 2022 Mayo | 38 | 64 | 102 |
| 2022 Abril | 42 | 34 | 76 |
| 2022 Marzo | 42 | 56 | 98 |
| 2022 Febrero | 44 | 30 | 74 |
| 2022 Enero | 51 | 37 | 88 |
| 2021 Diciembre | 50 | 30 | 80 |
| 2021 Noviembre | 38 | 48 | 86 |
| 2021 Octubre | 57 | 53 | 110 |
| 2021 Septiembre | 43 | 43 | 86 |
| 2021 Agosto | 45 | 26 | 71 |
| 2021 Julio | 53 | 35 | 88 |
| 2021 Junio | 25 | 41 | 66 |
| 2021 Mayo | 40 | 48 | 88 |
| 2021 Abril | 80 | 57 | 137 |
| 2021 Marzo | 65 | 38 | 103 |
| 2021 Febrero | 67 | 36 | 103 |
| 2021 Enero | 46 | 32 | 78 |
| 2020 Diciembre | 49 | 31 | 80 |
| 2020 Noviembre | 24 | 25 | 49 |
| 2020 Octubre | 27 | 15 | 42 |
| 2020 Septiembre | 33 | 25 | 58 |
| 2020 Agosto | 43 | 25 | 68 |
| 2020 Julio | 18 | 19 | 37 |
| 2020 Junio | 27 | 30 | 57 |
| 2020 Mayo | 17 | 9 | 26 |
| 2020 Abril | 20 | 14 | 34 |
| 2020 Marzo | 8 | 5 | 13 |
| 2020 Febrero | 2 | 2 | 4 |
| 2019 Mayo | 0 | 2 | 2 |
| 2019 Marzo | 5 | 0 | 5 |
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