Case for Diagnosis
Persistent Periorbital Edema
Edema periorbitario persistente
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A) Sección histológica (H-E, x40) en la que se aprecia, en el centro de la imagen, una célula gigante multinucleada con células muriformes o cuerpos de Medlar en su interior. B) Sección histológica (H-E, x63) en la que se distinguen muy bien los cuerpos de Medlar en el centro de la imagen. C) Sección histológica (PAS, x40), en la que se observan de forma detallada los cuerpos de Medlar. D) Sección histológica (metenamina de plata, x40), en la que también se observan de forma detallada los cuerpos de Medlar.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "J.F. Orts Paco, M.C. Soria Martínez, C. Godoy Alba, J. Navarro Pascual" "autores" => array:4 [ 0 => array:2 [ "nombre" => "J.F." "apellidos" => "Orts Paco" ] 1 => array:2 [ "nombre" => "M.C." "apellidos" => "Soria Martínez" ] 2 => array:2 [ "nombre" => "C." "apellidos" => "Godoy Alba" ] 3 => array:2 [ "nombre" => "J." "apellidos" => "Navarro Pascual" ] ] ] ] ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731022007645?idApp=UINPBA000044" "url" => "/00017310/0000011400000003/v1_202303051528/S0001731022007645/v1_202303051528/es/main.assets" ] "itemAnterior" => array:19 [ "pii" => "S0001731022007621" "issn" => "00017310" "doi" => "10.1016/j.ad.2021.08.016" "estado" => "S300" "fechaPublicacion" => "2023-03-01" "aid" => "3175" "copyright" => "AEDV" "documento" => "simple-article" "crossmark" => 1 "licencia" => "http://creativecommons.org/licenses/by-nc-nd/4.0/" "subdocumento" => "crp" "cita" => "Actas Dermosifiliogr. 2023;114:259-60" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "es" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">CASOS PARA EL DIAGNÓSTICO</span>" "titulo" => "Edema periorbitario persistente" "tienePdf" => "es" "tieneTextoCompleto" => "es" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "259" "paginaFinal" => "260" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Persistent periorbital edema" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figura 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 489 "Ancho" => 1255 "Tamanyo" => 190688 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Hematoxilina-eosina (H-E, ×10). Áreas de vacuolización de la capa basal junto con exocitosis de células inflamatorias. B) Tinción azul alcián (×10). Epidermis adelgazada e incremento de mucina en la dermis.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "V.H. Pinos León, J. Granizo Rubio, J. Sánchez Villarroel" "autores" => array:3 [ 0 => array:2 [ "nombre" => "V.H." "apellidos" => "Pinos León" ] 1 => array:2 [ "nombre" => "J." "apellidos" => "Granizo Rubio" ] 2 => array:2 [ "nombre" => "J." "apellidos" => "Sánchez Villarroel" ] ] ] ] ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731022007621?idApp=UINPBA000044" "url" => "/00017310/0000011400000003/v1_202303051528/S0001731022007621/v1_202303051528/es/main.assets" ] "asociados" => array:1 [ 0 => array:19 [ "pii" => "S0001731022007621" "issn" => "00017310" "doi" => "10.1016/j.ad.2021.08.016" "estado" => "S300" "fechaPublicacion" => "2023-03-01" "aid" => "3175" "copyright" => "AEDV" "documento" => "simple-article" "crossmark" => 1 "licencia" => "http://creativecommons.org/licenses/by-nc-nd/4.0/" "subdocumento" => "crp" "cita" => "Actas Dermosifiliogr. 2023;114:259-60" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "es" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">CASOS PARA EL DIAGNÓSTICO</span>" "titulo" => "Edema periorbitario persistente" "tienePdf" => "es" "tieneTextoCompleto" => "es" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "259" "paginaFinal" => "260" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Persistent periorbital edema" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figura 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 489 "Ancho" => 1255 "Tamanyo" => 190688 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Hematoxilina-eosina (H-E, ×10). Áreas de vacuolización de la capa basal junto con exocitosis de células inflamatorias. B) Tinción azul alcián (×10). Epidermis adelgazada e incremento de mucina en la dermis.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "V.H. Pinos León, J. Granizo Rubio, J. Sánchez Villarroel" "autores" => array:3 [ 0 => array:2 [ "nombre" => "V.H." "apellidos" => "Pinos León" ] 1 => array:2 [ "nombre" => "J." "apellidos" => "Granizo Rubio" ] 2 => array:2 [ "nombre" => "J." 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"apellidos" => "Pinos León" "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" ] ] ] 1 => array:3 [ "nombre" => "J." "apellidos" => "Granizo Rubio" "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:4 [ "nombre" => "J." "apellidos" => "Sanchez Villarroel" "email" => array:1 [ 0 => "j_marisolsv@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Metropolitano, Quito, Ecuador" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Universidad Central del Ecuador, Quito, Ecuador" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Edema periorbitario persistente" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1452 "Ancho" => 1083 "Tamanyo" => 130399 ] ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Medical History</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 57-year-old man with no disease history of interest consulted for episodes of palpebral edema, mainly affecting the right eye. The episodes had been occurring for 8 years. The edema spread to the rest of the face and was accompanied by moderately intense erythema. The clinical picture was not associated with a trigger, and no additional symptoms were reported.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">The physical examination revealed bilateral palpebral edema extending to the center of the face and cheeks and accompanied by erythema and scaling that spared the forehead and eyelids (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Additional Tests</span><p id="par0015" class="elsevierStylePara elsevierViewall">The blood workup, which included a complete blood count, liver function, kidney function, and autoimmune tests, revealed no significant abnormalities. The histopathology study revealed a thinned horny layer with areas of vacuolization in the basal layer that extended to the infundibular epithelium. The dermis was characterized by edema and an abundant lymphocytic infiltrate surrounding the hair follicles and blood vessels (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A). Alcian blue staining showed mucin to be increased in the dermis and around the adnexa (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">What is Your Diagnosis?</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Diagnosis</span><p id="par0025" class="elsevierStylePara elsevierViewall">Cutaneous lupus erythematosus.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Clinical Course and Treatment</span><p id="par0030" class="elsevierStylePara elsevierViewall">The patient was prescribed strict photoprotection, together with hydroxychloroquine 400<span class="elsevierStyleHsp" style=""></span>mg/d. Since he did not attend the subsequent checkups, the outcome remains unknown.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Comment</span><p id="par0035" class="elsevierStylePara elsevierViewall">Lupus erythematosus is an autoimmune disease of the connective tissue that usually progresses with a broad spectrum of clinical, histopathological, and immunological findings. The skin is usually one of the most affected organs.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The most appropriate classification for the wide range of lesions in lupus erythematosus continues to be debated. However, that of Gilliam and Sontheimer in 1981 is the most widely accepted to date.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Periorbital edema and erythema comprise a rare presentation of chronic cutaneous lupus erythematosus, whose pathophysiological mechanisms are not fully established. Several authors show that the development of edema is favored by the characteristic presence of lax connective tissue and local interstitial mucin deposits.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Similar findings were reported in the case in which the area affected was the glabella, from where the biopsy was taken.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Involvement of both the upper and lower eyelids has been reported. From the lower eyelid it can extend to the cheeks and may sometimes be accompanied by erythema and pruritus. According to a study by Wu et al.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> in 2018, this condition mainly affects middle-aged women, in whom unilateral left side involvement is the most common finding.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In some publications, these atypical lesions are classed within the spectrum of chronic cutaneous lupus, with the discoid variant being considered the most common, even though it does not have typical characteristics, such as atrophy, scarring, and follicular plugging.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> However, the persistent and progressive course of the palpebral and facial edema in these cases accounts for their inclusion in this subtype.</p><p id="par0060" class="elsevierStylePara elsevierViewall">As for serology findings, chronic cutaneous lupus is characterized by a lower incidence of positive autoantibodies, such as antinuclear antibodies, anti-double-stranded DNA antibodies, and anti-Sm antibodies. Such were the findings in the case we describe, as in most previously reported cases, where the peripheral blood autoimmunity workup also yielded negative results. Nevertheless, diagnosis is necessarily based on histopathological confirmation. The usual findings correspond to vacuolar degeneration of the basal layer together with epidermal atrophy and abnormalities of the dermis characterized by the presence of a dense perivascular and periadnexal infiltrate.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">The recurrence of symptoms despite multiple treatments and their exacerbation on exposure to sunlight enable us to rule out irritant contact dermatitis, cellulitis, and erysipelas. Heliotrope rash in dermatomyositis can be differentiated by its violaceous coloring, bilateral involvement, and the fact that it is accompanied by characteristic systemic and cutaneous manifestations. Telangiectasias, papules, and pustules (not reported in this manifestation of lupus erythematosus) may be found in Morbihan disease, which is considered a variant of rosacea and progresses with indurated edema affecting the upper two-thirds of the face and whose histopathological findings are nonspecific. Other differential diagnoses to be considered include sarcoidosis, foreign body granuloma, superior vena cava syndrome, and reactions to injectable fillers.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Treatment is based on photoprotection and topical therapy with corticosteroids or calcineurin inhibitors. However, antimalarial drugs continue to be the option of choice.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Finally, it is well known that some subtypes of lupus erythematosus progress with facial lesions. However, certain signs, such as chronic facial and palpebral edema, with no related findings, continue to be an uncommon and underdiagnosed manifestation, with few cases reported in the last few decades. Therefore, assessment of this condition should take into account a large number of differential diagnoses, among which cutaneous lupus erythematosus is a possibility that should always be investigated.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conflict of Interest</span><p id="par0080" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:2 [ "identificador" => "sec0040" "titulo" => "Medical History" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Physical Examination" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Additional Tests" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Diagnosis" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Clinical Course and Treatment" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Comment" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Conflict of Interest" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1452 "Ancho" => 1083 "Tamanyo" => 130399 ] ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 651 "Ancho" => 1667 "Tamanyo" => 297324 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Hematoxylin–eosin, ×10. Areas of vacuolization of the basal layer, together with exocytosis of inflammatory cells. (B) Alcian blue staining (×10). Thinned epidermis and increased mucin in the dermis.</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" => "Cutaneous lupus erythematosus: progress and challenges" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "A.J. Petty" 1 => "L. Floyd" 2 => "C. Henderson" 3 => "M.W. Nicholas" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s11882-020-0895-x" "Revista" => array:6 [ "tituloSerie" => "Curr Allergy Asthma Rep" "fecha" => "2020" "volumen" => "20" "paginaInicial" => "1" "paginaFinal" => "10" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31912246" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0040" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The classification and diagnosis of cutaneous lupus erythematosus" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "A. Kuhn" 1 => "A. 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