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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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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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 9 | 8 | 17 |
| 2024 October | 60 | 62 | 122 |
| 2024 September | 74 | 47 | 121 |
| 2024 August | 95 | 80 | 175 |
| 2024 July | 58 | 41 | 99 |
| 2024 June | 75 | 47 | 122 |
| 2024 May | 67 | 41 | 108 |
| 2024 April | 89 | 37 | 126 |
| 2024 March | 53 | 36 | 89 |
| 2024 February | 75 | 39 | 114 |
| 2024 January | 67 | 38 | 105 |
| 2023 December | 55 | 12 | 67 |
| 2023 November | 83 | 24 | 107 |
| 2023 October | 63 | 31 | 94 |
| 2023 September | 50 | 33 | 83 |
| 2023 August | 46 | 24 | 70 |
| 2023 July | 61 | 27 | 88 |
| 2023 June | 40 | 24 | 64 |
| 2023 May | 59 | 28 | 87 |
| 2023 April | 34 | 17 | 51 |
| 2023 March | 52 | 32 | 84 |
| 2023 February | 50 | 24 | 74 |
| 2023 January | 39 | 33 | 72 |
| 2022 December | 73 | 35 | 108 |
| 2022 November | 40 | 24 | 64 |
| 2022 October | 94 | 32 | 126 |
| 2022 September | 22 | 42 | 64 |
| 2022 August | 47 | 27 | 74 |
| 2022 July | 24 | 41 | 65 |
| 2022 June | 20 | 31 | 51 |
| 2022 May | 38 | 64 | 102 |
| 2022 April | 42 | 34 | 76 |
| 2022 March | 42 | 56 | 98 |
| 2022 February | 44 | 30 | 74 |
| 2022 January | 51 | 37 | 88 |
| 2021 December | 50 | 30 | 80 |
| 2021 November | 38 | 48 | 86 |
| 2021 October | 57 | 53 | 110 |
| 2021 September | 43 | 43 | 86 |
| 2021 August | 45 | 26 | 71 |
| 2021 July | 53 | 35 | 88 |
| 2021 June | 25 | 41 | 66 |
| 2021 May | 40 | 48 | 88 |
| 2021 April | 80 | 57 | 137 |
| 2021 March | 65 | 38 | 103 |
| 2021 February | 67 | 36 | 103 |
| 2021 January | 46 | 32 | 78 |
| 2020 December | 49 | 31 | 80 |
| 2020 November | 24 | 25 | 49 |
| 2020 October | 27 | 15 | 42 |
| 2020 September | 33 | 25 | 58 |
| 2020 August | 43 | 25 | 68 |
| 2020 July | 18 | 19 | 37 |
| 2020 June | 27 | 30 | 57 |
| 2020 May | 17 | 9 | 26 |
| 2020 April | 20 | 14 | 34 |
| 2020 March | 8 | 5 | 13 |
| 2020 February | 2 | 2 | 4 |
| 2019 May | 0 | 2 | 2 |
| 2019 March | 5 | 0 | 5 |
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