Case and Research Letter
Recurrent episodes of periorbital edema in an elderly woman
Edema recidivante periorbitario en una anciana
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Directora" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "704" "paginaFinal" => "706" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "R. Rodriguez-Lojo, I. Castiñeiras, M. Sánchez-Blas, M.L. Fernández-Diaz" "autores" => array:4 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Rodriguez-Lojo" "email" => array:1 [ 0 => "rodriguezlojo@hotmail.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" => "I." "apellidos" => "Castiñeiras" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "M." "apellidos" => "Sánchez-Blas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "M.L." "apellidos" => "Fernández-Diaz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Dermatología Hospital Universitário Lucus Augusti, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Anatomía Patológica, Hospital Universitário Lucus Augusti, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Edema recidivante periorbitario en una anciana" ] ] "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" => 629 "Ancho" => 800 "Tamanyo" => 90272 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Edematous plaque on the right periocular area.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">An 81-year-old woman was referred for an edematous, erythematous plaque that had arisen 48<span class="elsevierStyleHsp" style=""></span>h earlier in the right periorbital region and on the ipsilateral side of the face. The lesions, present on both cheeks, initially had a papular-vesicular appearance, but they subsequently increased in size and became firm and tender and evolved rapidly into edematous plaques. The patient reported pruritus and fever of 38<span class="elsevierStyleHsp" style=""></span>°C. There was no history of insect bite, trauma, foreign travel, or gastrointestinal problems in the previous months. The patient was not taking any regular medication, including topical medication, at the time of onset of the lesions, nor did she use cosmetic preparations or contact lenses.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Three years earlier she had attended the infectious diseases department with a sore, pruritic erythematous plaque affecting the whole left side of the face, associated with fever. Antimicrobial therapy with intravenous amoxicillin plus clavulanic acid had been administered and the lesion resolved within a week, without scarring. A diagnosis of a probable acute bacterial cellulitis had been made. There was no other medical history of note and her family history was unremarkable.</p><p id="par0015" class="elsevierStylePara elsevierViewall">On physical examination, the patient had a healthy appearance, was afebrile, and presented a tender, infiltrated erythematous plaque that affected the periorbital region and cheek on the right side of the face (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). There were no palpable lymph nodes and no other skin lesions. The total IgE concentration was 1334<span class="elsevierStyleHsp" style=""></span>kU/L (normal range, 0–100<span class="elsevierStyleHsp" style=""></span>kU/L). Complete blood count, biochemistry, and serologic tests for antinuclear antibodies, antineutrophil cytoplasmic antibodies, and C1 inhibitor and C4 levels were normal or negative. Chest and sinus X-rays were normal. Facial computed tomography showed no ocular or intracranial alterations. Ophthalmologic examination was normal in both eyes and no ocular dysfunction was detected. Direct immunofluorescence for herpes viruses was negative.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The suspected clinical diagnosis was an insect bite reaction with acute cellulitis. The patient was treated with intravenous cloxacillin and the skin lesions improved within a few days.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Two weeks later, the patient had a recurrence, with the reappearance of an erythematous, edematous plaque in the right periocular area, associated with bullous papules on both arms and hands. These lesions were initially bright red, but progressively darkened to a violaceous color, giving the appearance of hematomas (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). There was no malaise or fever. Laboratory studies were within normal limits, including the peripheral eosinophil count. The total IgE concentration was 910<span class="elsevierStyleHsp" style=""></span>kU/L.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Histopathology study of a skin biopsy taken from the left wrist revealed marked edema of the papillary dermis with a moderately dense superficial and deep perivascular and interstitial infiltrate of eosinophils and lymphocytes throughout the dermis and subcutaneous fat. Additionally, degranulated material and fragmented eosinophils were observed around collagen fibers in the dermis and were identified as flame figures (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Direct immunofluorescence was negative. A diagnosis of Wells syndrome was made.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Oral steroid therapy was started at 0.5<span class="elsevierStyleHsp" style=""></span>mg/kg/d and the patient's cutaneous signs and symptoms improved within 24<span class="elsevierStyleHsp" style=""></span>h, leaving residual lesions. The dose of oral prednisone was tapered over 6 days. However, 10 days after withdrawal of the corticosteroid treatment, the patient presented recurrence in the form of edema in the left periorbital region and vesicles on forehead; there were no systemic symptoms at that time. A further cycle of oral steroids (prednisone 30<span class="elsevierStyleHsp" style=""></span>mg/d) was administered, leading to a notable improvement, but a new flare-up occurred when the dose was reduced below 10<span class="elsevierStyleHsp" style=""></span>mg a day. Oral prednisone, 10<span class="elsevierStyleHsp" style=""></span>mg/d, was therefore continued for a month, with no adverse effects. At the end of that cycle, the lesions had cleared completely and the patient has subsequently remained symptom-free.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Wells syndrome is a rare dermatosis of unknown etiology and pathogenesis. It is typically characterized by recurrent episodes of pruritic plaques, occasionally associated with bullae, which develop over 2–3 days and usually resolve spontaneously over 2–8 weeks. The site and appearance of the skin lesions vary, but they often affect the limbs. Facial lesions are rare. In our patient, the periorbital region was the most severely affected area, alternating from right to left sides in successive recurrences.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1–3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The most common systemic complaint is malaise. Fever is present in less than a quarter of cases. The manifestations of eosinophilic cellulitis are difficult to differentiate from bacterial cellulitis, and misdiagnosis is common. In our case, the first skin lesions were associated with fever, leading to a misdiagnosis of bacterial cellulitis. Although there was an initial improvement with antimicrobial therapy, we consider this as a coincidence and that the cutaneous lesions had probably undergone spontaneous remission.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Approximately 50% of cases reported in the literature have presented peripheral blood eosinophilia during active disease, but the eosinophil count remained normal in our patient.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">4–8</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion, we have described a patient initially diagnosed with bacterial cellulitis, but in whom skin biopsy subsequently established a diagnosis of eosinophilic cellulitis. This occurred in the absence of any obvious trigger or associated skin conditions and showed a good response to steroid therapy. We would like to draw attention to the atypical periorbital site of the lesions.</p><p id="par0060" class="elsevierStylePara elsevierViewall">We consider eosinophilic cellulitis should be included in the differential diagnosis of recurrent episodes of edema in the periorbital region (<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>).<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a></p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical disclosures</span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Protection of human and animal subjects</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this investigation.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Confidentiality of data</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appears in this article.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Right to privacy and informed consent</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appears in this article.</p></span></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Conflict of interests</span><p id="par0080" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:3 [ "identificador" => "sec0005" "titulo" => "Ethical disclosures" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0010" "titulo" => "Protection of human and animal subjects" ] 1 => array:2 [ "identificador" => "sec0015" "titulo" => "Confidentiality of data" ] 2 => array:2 [ "identificador" => "sec0020" "titulo" => "Right to privacy and informed consent" ] ] ] 1 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflict of interests" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:4 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 629 "Ancho" => 800 "Tamanyo" => 90272 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Edematous plaque on the right periocular area.</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" => 600 "Ancho" => 800 "Tamanyo" => 68157 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Violaceous plaque on finger.</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" => 880 "Ancho" => 1600 "Tamanyo" => 404084 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Infiltrate of eosinophils in the fat and flame figure in the dermis.</p>" ] ] 3 => array:8 [ "identificador" => "tbl0005" "etiqueta" => "Table 1" "tipo" => "MULTIMEDIATABLA" "mostrarFloat" => true "mostrarDisplay" => false "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at1" "detalle" => "Table " "rol" => "short" ] ] "tabla" => array:2 [ "tablatextoimagen" => array:1 [ 0 => array:2 [ "tabla" => array:1 [ 0 => """ <table border="0" frame="\n \t\t\t\t\tvoid\n \t\t\t\t" class=""><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Infectious diseases \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">Cellulitis, erysipelas, herpesvirus infection trichinosis, rickettsiosis, anthrax, fungal infections, tungiasis, tularemia \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Inflammatory and autoimmune diseases \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">Angioedema, urticaria<br>Rosacea<br>Allergic/irritant contact dermatitis<br>Sweet syndrome<br>Eosinophilic dermatoses<br>Dermatomyositis, lupus erythematosus \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Systemic diseases \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">Thyroid, kidney, liver, and heart diseases \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Drugs and therapies \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">Imatinib, rifampicin, niacin, clozapine, ipilimumab, risperidone, aspirin, irbersartan, chlorpromazine<br>Positive airway pressure ventilation \t\t\t\t\t\t\n \t\t\t\t</td></tr></tbody></table> """ ] "imagenFichero" => array:1 [ 0 => "xTab1390657.png" ] ] ] "notaPie" => array:1 [ 0 => array:3 [ "identificador" => "tblfn0005" "etiqueta" => "a" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Adapted from Veraldi et al.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a></p>" ] ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Differential diagnosis of recurrent periorbital edema.<a class="elsevierStyleCrossRef" href="#tblfn0005"><span class="elsevierStyleSup">a</span></a></p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Successful treatment of steroid-dependent eosinophilic cellulitis with cyclosporine" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "S.H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 7 | 13 | 20 |
| 2024 October | 81 | 60 | 141 |
| 2024 September | 89 | 24 | 113 |
| 2024 August | 125 | 53 | 178 |
| 2024 July | 88 | 32 | 120 |
| 2024 June | 96 | 37 | 133 |
| 2024 May | 94 | 40 | 134 |
| 2024 April | 71 | 27 | 98 |
| 2024 March | 90 | 30 | 120 |
| 2024 February | 66 | 23 | 89 |
| 2024 January | 55 | 25 | 80 |
| 2023 December | 61 | 31 | 92 |
| 2023 November | 63 | 28 | 91 |
| 2023 October | 54 | 20 | 74 |
| 2023 September | 106 | 28 | 134 |
| 2023 August | 52 | 13 | 65 |
| 2023 July | 80 | 35 | 115 |
| 2023 June | 55 | 25 | 80 |
| 2023 May | 91 | 32 | 123 |
| 2023 April | 75 | 14 | 89 |
| 2023 March | 73 | 21 | 94 |
| 2023 February | 47 | 24 | 71 |
| 2023 January | 38 | 38 | 76 |
| 2022 December | 67 | 49 | 116 |
| 2022 November | 29 | 20 | 49 |
| 2022 October | 25 | 26 | 51 |
| 2022 September | 42 | 26 | 68 |
| 2022 August | 19 | 29 | 48 |
| 2022 July | 25 | 33 | 58 |
| 2022 June | 25 | 22 | 47 |
| 2022 May | 91 | 36 | 127 |
| 2022 April | 64 | 27 | 91 |
| 2022 March | 85 | 43 | 128 |
| 2022 February | 56 | 16 | 72 |
| 2022 January | 81 | 30 | 111 |
| 2021 December | 49 | 27 | 76 |
| 2021 November | 44 | 40 | 84 |
| 2021 October | 64 | 45 | 109 |
| 2021 September | 41 | 40 | 81 |
| 2021 August | 77 | 28 | 105 |
| 2021 July | 66 | 21 | 87 |
| 2021 June | 56 | 15 | 71 |
| 2021 May | 49 | 37 | 86 |
| 2021 April | 248 | 65 | 313 |
| 2021 March | 60 | 26 | 86 |
| 2021 February | 55 | 25 | 80 |
| 2021 January | 29 | 29 | 58 |
| 2020 December | 39 | 16 | 55 |
| 2020 November | 25 | 20 | 45 |
| 2020 October | 32 | 9 | 41 |
| 2020 September | 39 | 11 | 50 |
| 2020 August | 20 | 12 | 32 |
| 2020 July | 16 | 14 | 30 |
| 2020 June | 25 | 20 | 45 |
| 2020 May | 15 | 18 | 33 |
| 2020 April | 13 | 17 | 30 |
| 2020 March | 14 | 12 | 26 |
| 2020 February | 1 | 1 | 2 |
| 2020 January | 11 | 2 | 13 |
| 2019 December | 2 | 0 | 2 |
| 2019 September | 4 | 0 | 4 |
| 2019 June | 2 | 0 | 2 |
| 2019 April | 4 | 4 | 8 |
| 2019 March | 2 | 0 | 2 |
| 2019 January | 1 | 0 | 1 |
| 2018 December | 2 | 0 | 2 |
| 2018 November | 3 | 0 | 3 |
| 2018 October | 1 | 0 | 1 |
| 2018 September | 3 | 0 | 3 |
| 2018 June | 0 | 1 | 1 |
| 2018 February | 19 | 6 | 25 |
| 2018 January | 29 | 6 | 35 |
| 2017 December | 41 | 7 | 48 |
| 2017 November | 31 | 9 | 40 |
| 2017 October | 43 | 8 | 51 |
| 2017 September | 28 | 12 | 40 |
| 2017 August | 20 | 5 | 25 |
| 2017 July | 30 | 4 | 34 |
| 2017 June | 24 | 5 | 29 |
| 2017 May | 25 | 4 | 29 |
| 2017 April | 14 | 4 | 18 |
| 2017 March | 15 | 7 | 22 |
| 2017 February | 37 | 7 | 44 |
| 2017 January | 15 | 13 | 28 |
| 2016 December | 25 | 14 | 39 |
| 2016 November | 47 | 19 | 66 |
| 2016 October | 112 | 24 | 136 |
| 2016 September | 2 | 0 | 2 |
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