Case Letter
PASH syndrome: a novel surgical approach
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true "cabecera" => "<span class="elsevierStyleTextfn">Case Letter</span>" "titulo" => "PASH syndrome: a novel surgical approach" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "121" "paginaFinal" => "123" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Irina Cawen, Jorge Navarrete, Caroline Agorio" "autores" => array:3 [ 0 => array:3 [ "nombre" => "Irina" "apellidos" => "Cawen" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:4 [ "nombre" => "Jorge" "apellidos" => "Navarrete" "email" => array:1 [ 0 => "jnavarrete90@gmail.com" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 2 => array:3 [ "nombre" => "Caroline" "apellidos" => "Agorio" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Department of Dermatology, Hospital de Clínicas “Dr. Manuel Quintela”, Montevideo, Uruguay" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Dermatology, Hospital Padre Hurtado, Santiago, Chile" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1883 "Ancho" => 2507 "Tamanyo" => 519659 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hidradenitis suppurativa of (A), left and (B), right axillae before surgical intervention. (C), Sacrococcygeal pilonidal cyst. (D), Ice pick scar from severe acne.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">PASH syndrome is a rare autoinflammatory syndrome (AIS), which consists of pyoderma gangrenosum (PG), acne conglobata and hidradenitis suppurativa (HS).<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> No standard treatment has been determined, although case reports have focused on systemic antibiotics, immunosuppressants and biologics.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A 45-year-old male presented with a 20-year history of HS on axillae and the inguinal region (Hurley stage III) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B). He had diabetes in treatment with metformin, and chronic hepatitis B infection. A sacrococcygeal pilonidal cyst was present (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C), as well as ice pick scars (from acne conglobata in his adolescence) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D). Treatment for his HS with multiple systemic antibiotics had failed. Immunosuppressants/biologics were contraindicated due to hepatitis B infection. Wide surgical excision was performed under local anesthesia (axillae and inguinal area, sequentially), with the approximation of borders and secondary intention healing (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The patient was instructed to maintain abduction.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Six weeks after surgery, he developed a rapidly growing lesion on his left leg (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>A), with intense pain, but no systemic involvement. This was interpreted as a bacterial skin infection or possibly PG, and empirical intravenous broad-spectrum antibiotics were initiated (penicillin, clindamycin, and ciprofloxacin for ten days), with clobetasol 0.05% ointment. He showed a good response, with cribriform scarring (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>B). Histopathology showed a dense neutrophilic infiltrate. Cultures were consistently negative. Diagnosis of PG was made, and we were able to categorize our case as PASH syndrome. Lab exams (CBC, ESR, liver and kidney function tests), colonoscopy, and upper endoscopy were normal.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Sequence analysis and deletion/duplication testing of genes LPIN2, MEFV, MVK, NLRP3, PSTPIP-1, and TNFRSF1A showed no pathogenic variants.</p><p id="par0025" class="elsevierStylePara elsevierViewall">With a 36-month follow-up, the patient has had no recurrence of PG or HS.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The diseases that constitute PASH syndrome seem to have independent temporal behaviors and responses to treatment. There are 31 other PASH cases in the available literature, of which 29 have a genetic study, showing heterogenic mutations (previously found in other AIS) in eight cases (8/29; 27.6%).</p><p id="par0035" class="elsevierStylePara elsevierViewall">Pilonidal cysts have not been described in PASH syndrome, although they are part of the follicular occlusion tetrad (along with dissecting cellulitis).</p><p id="par0040" class="elsevierStylePara elsevierViewall">While systemic corticosteroids are the treatment of choice for PG, systemic antibiotics and topical corticosteroids are feasible therapeutic options in diabetic patients.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Given our good experience with surgery on non-PASH HS, as well as in other centers, we chose this strategy, and highlight the good results obtained, something not previously described for PASH syndrome.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> This also spares the patient from immunosuppression and other adverse effects of biologics, at a lower cost. The risk of pathergy is a limitation. We recommend three intradermal 20-G needle pricks on the ventral forearm and evaluate at 48 hours in patients with HS and severe acne before surgery is considered since they might represent a case of latent PASH syndrome.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">A deeper knowledge of etiopathogenesis may lead to targeted therapeutic strategies, however, this will certainly pose a challenge in a disease as polymorphic as PASH. Treatment must therefore be individualized and considering surgery showed promising results, we believe it merits further research.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0055" class="elsevierStylePara elsevierViewall">The <span class="elsevierStyleGrantSponsor" id="gs0005"><span class="elsevierStyleItalic">Fundación Manuel Quintela</span></span> helped finance the genetic testing.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors' contributions</span><p id="par0060" class="elsevierStylePara elsevierViewall">Irina Cawen: Approval of the final version of the manuscript; critical literature review; data collection, analysis, and interpretation; effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic; management of studied cases; manuscript critical review; preparation and writing of the manuscript; statistical analysis; study conception and planning.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Jorge Navarrete: Approval of the final version of the manuscript; critical literature review; data collection, analysis, and interpretation; effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic; management of studied cases; manuscript critical review; preparation and writing of the manuscript; statistical analysis; study conception and planning.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Caroline Agorio: Approval of the final version of the manuscript; critical literature review; effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic; management of studied cases; manuscript critical review; study conception and planning.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Authors' contributions" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-07-22" "fechaAceptado" => "2020-08-24" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Department of Dermatology, Hospital de Clínicas “Dr. Manuel Quint<span class="elsevierStyleItalic">ela</span>”, Montevideo, Uruguay.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1883 "Ancho" => 2507 "Tamanyo" => 519659 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hidradenitis suppurativa of (A), left and (B), right axillae before surgical intervention. (C), Sacrococcygeal pilonidal cyst. (D), Ice pick scar from severe acne.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 2483 "Ancho" => 1674 "Tamanyo" => 521270 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A), Wide surgical excision of hidradenitis suppurativa on right axilla, with approximation of borders and (B), secondary intention healing with calcium alginate followed by hydrocolloid dressings at 36 weeks, with certain retraction, but no functional impairment.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1791 "Ancho" => 1674 "Tamanyo" => 306515 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">(A), Pyoderma gangrenosum (erythematous lesion with blisters, pustules, and a purplish hue) on his left leg before intravenous broad-spectrum antibiotics and topical high-potency corticosteroids. (B), One month after treatment; cribriform scarring can be seen.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pyoderma gangrenosum, acne, and suppurative hidradenitis (PASH) – a new autoinflammatory syndrome distinct from PAPA syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M. Braun-Falco" 1 => "O. Kovnerystyy" 2 => "P. Lohse" 3 => "T. Ruzicka" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.jaad.2010.12.025" "Revista" => array:6 [ "tituloSerie" => "J Am Acad Dermatol." 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| Ano/Mês | Html | Total | |
|---|---|---|---|
| 2024 Novembro | 24 | 8 | 32 |
| 2024 Outubro | 162 | 91 | 253 |
| 2024 Setembro | 178 | 105 | 283 |
| 2024 Agosto | 206 | 128 | 334 |
| 2024 Julho | 178 | 124 | 302 |
| 2024 Junho | 157 | 88 | 245 |
| 2024 Maio | 112 | 77 | 189 |
| 2024 Abril | 180 | 101 | 281 |
| 2024 Março | 179 | 84 | 263 |
| 2024 Fevereiro | 221 | 88 | 309 |
| 2024 Janeiro | 162 | 61 | 223 |
| 2023 Dezembro | 200 | 63 | 263 |
| 2023 Novembro | 232 | 100 | 332 |
| 2023 Outubro | 170 | 84 | 254 |
| 2023 Setembro | 159 | 89 | 248 |
| 2023 Agosto | 145 | 44 | 189 |
| 2023 Julho | 101 | 44 | 145 |
| 2023 Junho | 104 | 46 | 150 |
| 2023 Maio | 137 | 48 | 185 |
| 2023 Abril | 120 | 19 | 139 |
| 2023 Março | 81 | 64 | 145 |
| 2023 Fevereiro | 50 | 28 | 78 |
| 2023 Janeiro | 81 | 63 | 144 |
| 2022 Dezembro | 79 | 36 | 115 |
| 2022 Novembro | 88 | 58 | 146 |
| 2022 Outubro | 116 | 60 | 176 |
| 2022 Setembro | 83 | 77 | 160 |
| 2022 Agosto | 48 | 59 | 107 |
| 2022 Julho | 58 | 69 | 127 |
| 2022 Junho | 67 | 67 | 134 |
| 2022 Maio | 60 | 60 | 120 |
| 2022 Abril | 76 | 68 | 144 |
| 2022 Março | 98 | 81 | 179 |
| 2022 Fevereiro | 98 | 65 | 163 |
| 2022 Janeiro | 126 | 86 | 212 |
| 2021 Dezembro | 49 | 64 | 113 |
| 2021 Novembro | 39 | 28 | 67 |
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