Case and Research Letter
A Case of Pyoderma Gangrenosum-Like Ulcers Progressing into Neutrophilic Panniculitis in a Patient With Myelodysplastic Syndrome
Un caso de úlceras similares al pioderma gangrenoso que posteriormente desarrollaron paniculitis neutrofílica en un paciente con síndrome mielodisplásico
Department of Dermatology, Fukushima Medical University, Fukushima, Japan
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Visión lateral izquierda. Gran placa eritematosa que ocupa la totalidad del muslo izquierdo y flanco ipsilateral. Placa bien delimitada, tumefacta y caliente. B: Imagen clínica. Visión posterior. Placa eritemato-edematosa. Afectación de muslo, glúteo y flanco izquierdos; confluyendo hacia la región lumbosacra. C: Imagen histológica. Panorámica. Piel con inflamación linfocitaria perivascular superficial con presencia de neutrófilos. Discreto edema dermis papilar y respeto epidérmico. No se observan imágenes de paniculitis ni vasculitis. No se aprecian eosinófilos. Imagen original 4 X. Tinción de hematoxilina-eosina. D: Imagen histológica. Detalle del infiltrado inflamatorio. Dermis papilar con infiltrado linfocitario perivascular. Presencia de neutrófilos. Imagen original 20 X. Tinción de hematoxilina-eosina. E: Imagen histológica. Detalle del infiltrado perianexial. Infiltrado inflamatorio perianexial compuesto por linfocitos y neutrófilos. Imagen original 20 X. Tinción de hematoxilina-eosina.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "K. Díez-Madueño, D. Buendía Castaño, D. Roldán Cortés, P. de la Cueva Dobao" "autores" => array:4 [ 0 => array:2 [ "nombre" => "K." "apellidos" => "Díez-Madueño" ] 1 => array:2 [ "nombre" => "D." "apellidos" => "Buendía Castaño" ] 2 => array:2 [ "nombre" => "D." "apellidos" => "Roldán Cortés" ] 3 => array:2 [ "nombre" => "P." 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Note the improvement of the lesions after 6 months of treatment with apremilast 30<span class="elsevierStyleHsp" style=""></span>mg twice a day (C and D).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M. Mansilla-Polo, C. Abril-Pérez, M.Á. Navarro-Mira, R. Botella-Estrada" "autores" => array:4 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "Mansilla-Polo" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Abril-Pérez" ] 2 => array:2 [ "nombre" => "M.Á." "apellidos" => "Navarro-Mira" ] 3 => array:2 [ "nombre" => "R." "apellidos" => "Botella-Estrada" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731023004908?idApp=UINPBA000044" "url" => "/00017310/unassign/S0001731023004908/v1_202307180913/en/main.assets" ] "asociados" => array:1 [ 0 => array:17 [ "pii" => "S0001731024006288" "issn" => "00017310" "doi" => "10.1016/j.ad.2024.07.016" "estado" => "S200" "fechaPublicacion" => "2024-08-16" "aid" => "4038" "copyright" => "AEDV" "documento" => "simple-article" "crossmark" => 0 "subdocumento" => "crp" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "es" => array:10 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Carta científico-clínica</span>" "titulo" => " Un caso de úlceras similares al pioderma gangrenoso que posteriormente desarrollaron paniculitis neutrofílica en un paciente con síndrome mielodisplásico" "tienePdf" => "es" "tieneTextoCompleto" => "es" "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "A Case of Pyoderma Gangrenosum-Like Ulcers Progressing into Neutrophilic Panniculitis in a Patient With Myelodysplastic Syndrome" ] ] "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" => 1286 "Ancho" => 2340 "Tamanyo" => 600780 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) Úlcera profunda bien demarcada en el sitio de inserción del catéter venoso central. (b) Nódulos eritematosos dolorosos en el antebrazo (flecha). (c) Lesión ulcerativa en la zona sacra. (d) Examen histopatológico que refleja paniculitis lobular (H-E, ×20). (e) Mayor amplificación que muestra infiltración neutrofílica en el tejido graso (H-E, ×200).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "K. Irie, T. Yamamoto" "autores" => array:2 [ 0 => array:2 [ "nombre" => "K." "apellidos" => "Irie" ] 1 => array:2 [ "nombre" => "T." "apellidos" => "Yamamoto" ] ] ] ] ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731024006288?idApp=UINPBA000044" "url" => "/00017310/unassign/S0001731024006288/v1_202408160426/es/main.assets" ] ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letter</span>" "titulo" => "A Case of Pyoderma Gangrenosum-Like Ulcers Progressing into Neutrophilic Panniculitis in a Patient With Myelodysplastic Syndrome" "tieneTextoCompleto" => true "saludo" => "To the Editor:" "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "K. Irie, T. Yamamoto" "autores" => array:2 [ 0 => array:4 [ "nombre" => "K." "apellidos" => "Irie" "email" => array:1 [ 0 => "kinuko07@fmu.ac.jp" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "T." "apellidos" => "Yamamoto" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology, Fukushima Medical University, Fukushima, Japan" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Un caso de úlceras similares al pioderma gangrenoso que posteriormente desarrollaron paniculitis neutrofílica en un paciente con síndrome mielodisplásico" ] ] "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" => 1286 "Ancho" => 2340 "Tamanyo" => 602804 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) Well-demarcated deep ulcer on the central vein catheter insertion site. (b) Painful erythematous nodules on the forearm (arrow). (c) Ulcerative lesion on the sacral area. (d) Histopathology examination showing lobular panniculitis (H–E, ×20). (e) Higher magnification showing neutrophil infiltration inside the fat tissue (H–E, ×200).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">This is a rare case that presented with pyoderma gangrenosum-like cervical ulcers followed by painful subcutaneous nodules on the lower legs showing neutrophilic panniculitis on the histopathological examination in a patient with myelodysplastic syndrome.</p><p id="par0010" class="elsevierStylePara elsevierViewall">This is the case of a 51-year-old man hospitalized due to myelodysplastic syndrome who received chemotherapy with azacytidine without granulocyte colony-stimulating factor. He developed a skin ulcer and subcutaneous induration at the central venous catheter insertion site on the right neck (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>a). Although skin biopsy was not performed at this time, PG was suspected, and oral prednisolone (PSL) (60<span class="elsevierStyleHsp" style=""></span>mg/day) was started. The skin ulcer healed in 3 weeks. After a remission induction therapy, anthracycline was administered for 3 days and cytarabine for 7, PSL was down-titrated to 25<span class="elsevierStyleHsp" style=""></span>mg, and no recurrences of the cervical lesions were seen. However, new and painful subcutaneous indurations appeared on the extremities with high fever. The blood test showed elevated C-reactive protein levels (2.51<span class="elsevierStyleHsp" style=""></span>mg/dL) and decreased platelet count (1.0<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">4</span>/μL). The physical examination revealed the presence of multiple tender subcutaneous erythematous indurations (about 0.5–2<span class="elsevierStyleHsp" style=""></span>cm in diameter), on the forearms and thighs, as well as sacral ulcer (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>b, c). Local samples for bacteriological and mycological tests tested negative, and the patient's response to antibiotics was poor. Skin biopsy of the subcutaneous induration on the left forearm showed hemorrhage, neutrophil and lymphocyte infiltration of the deep dermis, and neutrophilic lobular panniculitis, without any signs of vasculitis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>d, e). The lesions on the extremities and sacral area improved without up-titration of the dose of PSL.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In the current case, deep ulcers at the central venous catheter insertion site on the right neck were considered a pathergy reaction, a characteristic feature of PG. There were no ulcerative lesions suggestive of PG on any part of the body other than the neck. After 2 months, painful subcutaneous erythematous nodules appeared on the extremities, and a skin biopsy revealed the presence of neutrophilic lobular panniculitis. Because neither septal panniculitis nor Miescher radial granuloma was seen, erythema nodosum was excluded. PG and neutrophilic panniculitis are both categorized as deep neutrophilic dermatoses,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> suggesting a shared pathogenesis. Neutrophilic lobular panniculitis is observed in subcutaneous Sweet's syndrome. PG and Sweet's syndrome are the representative diseases of neutrophilic dermatosis, and both diseases often clinically present with similar features, which are difficult to differentiate, especially in patients with hematologic disorders.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2–4</span></a> Such overlapped or atypical cases have been reported under the term ‘neutrophilic dermatosis of myeloproliferative disorders’, suggesting a linking of entities of both disorders in a continuous pathologic spectrum.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> Although the cervical ulcer was not biopsized in the present case, ulcerative neutrophilic panniculitis cannot be ruled out.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The VEXAS syndrome (vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic) is an inflammatory disease due to somatic UBA1 variants presenting with myelodysplasia, and autoinflammatory symptoms including chondritis, vasculitis, and neutrophilic dermatosis.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> The VEXAS syndrome was listed as a differential disease because of the common complication of neutrophilic dermatosis and myelodysplastic syndrome. However, it was ruled out in this case because the granulocyte progenitor cells did not contain cytoplasmic vacuoles in the bone marrow biopsy. The current case of myelodysplastic syndrome presented with different clinical phenotypes such as deep ulcer, and painful erythematous subcutaneous nodules at different points in time, both of which were due to activated neutrophils.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interests" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1286 "Ancho" => 2340 "Tamanyo" => 602804 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) Well-demarcated deep ulcer on the central vein catheter insertion site. (b) Painful erythematous nodules on the forearm (arrow). (c) Ulcerative lesion on the sacral area. (d) Histopathology examination showing lobular panniculitis (H–E, ×20). (e) Higher magnification showing neutrophil infiltration inside the fat tissue (H–E, ×200).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "From acute febrile neutrophilic dermatosis to neutrophilic disease: forty years of clinical research" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "D. 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Pei" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "N Engl J Med" "fecha" => "2020" "volumen" => "31" "paginaInicial" => "2628" "paginaFinal" => "2638" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/00017310/unassign/S0001731023009262/v1_202401211204/en/main.assets" "Apartado" => null "PDF" => "https://static.elsevier.es/multimedia/00017310/unassign/S0001731023009262/v1_202401211204/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731023009262?idApp=UINPBA000044" ]
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