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pruritic erythematous plaque affecting the whole left side of the face&#44; associated with fever&#46; Antimicrobial therapy with intravenous amoxicillin plus clavulanic acid had been administered and the lesion resolved within a week&#44; without scarring&#46; A diagnosis of a probable acute bacterial cellulitis had been made&#46; There was no other medical history of note and her family history was unremarkable&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">On physical examination&#44; the patient had a healthy appearance&#44; was afebrile&#44; and presented a tender&#44; infiltrated erythematous plaque that affected the periorbital region and cheek on the right side of the face &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; There were no palpable lymph nodes and no other skin lesions&#46; The total IgE concentration was 1334<span class="elsevierStyleHsp" style=""></span>kU&#47;L &#40;normal range&#44; 0&#8211;100<span class="elsevierStyleHsp" style=""></span>kU&#47;L&#41;&#46; Complete blood count&#44; biochemistry&#44; and serologic tests for antinuclear antibodies&#44; antineutrophil cytoplasmic antibodies&#44; and C1 inhibitor and C4 levels were normal or negative&#46; Chest and sinus X-rays were normal&#46; Facial computed tomography showed no ocular or intracranial alterations&#46; Ophthalmologic examination was normal in both eyes and no ocular dysfunction was detected&#46; Direct immunofluorescence for herpes viruses was negative&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The suspected clinical diagnosis was an insect bite reaction with acute cellulitis&#46; The patient was treated with intravenous cloxacillin and the skin lesions improved within a few days&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Two weeks later&#44; the patient had a recurrence&#44; with the reappearance of an erythematous&#44; edematous plaque in the right periocular area&#44; associated with bullous papules on both arms and hands&#46; These lesions were initially bright red&#44; but progressively darkened to a violaceous color&#44; giving the appearance of hematomas &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; There was no malaise or fever&#46; Laboratory studies were within normal limits&#44; including the peripheral eosinophil count&#46; The total IgE concentration was 910<span class="elsevierStyleHsp" style=""></span>kU&#47;L&#46;</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&#46; Additionally&#44; degranulated material and fragmented eosinophils were observed around collagen fibers in the dermis and were identified as flame figures &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Direct immunofluorescence was negative&#46; A diagnosis of Wells syndrome was made&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Oral steroid therapy was started at 0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;d and the patient&#39;s cutaneous signs and symptoms improved within 24<span class="elsevierStyleHsp" style=""></span>h&#44; leaving residual lesions&#46; The dose of oral prednisone was tapered over 6 days&#46; However&#44; 10 days after withdrawal of the corticosteroid treatment&#44; the patient presented recurrence in the form of edema in the left periorbital region and vesicles on forehead&#59; there were no systemic symptoms at that time&#46; A further cycle of oral steroids &#40;prednisone 30<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41; was administered&#44; leading to a notable improvement&#44; but a new flare-up occurred when the dose was reduced below 10<span class="elsevierStyleHsp" style=""></span>mg a day&#46; Oral prednisone&#44; 10<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#44; was therefore continued for a month&#44; with no adverse effects&#46; At the end of that cycle&#44; the lesions had cleared completely and the patient has subsequently remained symptom-free&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Wells syndrome is a rare dermatosis of unknown etiology and pathogenesis&#46; It is typically characterized by recurrent episodes of pruritic plaques&#44; occasionally associated with bullae&#44; which develop over 2&#8211;3 days and usually resolve spontaneously over 2&#8211;8 weeks&#46; The site and appearance of the skin lesions vary&#44; but they often affect the limbs&#46; Facial lesions are rare&#46; In our patient&#44; the periorbital region was the most severely affected area&#44; alternating from right to left sides in successive recurrences&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The most common systemic complaint is malaise&#46; Fever is present in less than a quarter of cases&#46; The manifestations of eosinophilic cellulitis are difficult to differentiate from bacterial cellulitis&#44; and misdiagnosis is common&#46; In our case&#44; the first skin lesions were associated with fever&#44; leading to a misdiagnosis of bacterial cellulitis&#46; Although there was an initial improvement with antimicrobial therapy&#44; we consider this as a coincidence and that the cutaneous lesions had probably undergone spontaneous remission&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Approximately 50&#37; of cases reported in the literature have presented peripheral blood eosinophilia during active disease&#44; but the eosinophil count remained normal in our patient&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">4&#8211;8</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion&#44; we have described a patient initially diagnosed with bacterial cellulitis&#44; 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Case and Research Letter
Recurrent episodes of periorbital edema in an elderly woman
Edema recidivante periorbitario en una anciana
R. Rodriguez-Lojoa,
Autor para correspondencia
rodriguezlojo@hotmail.com

Corresponding author.
, I. Castiñeirasa, M. Sánchez-Blasb, M.L. Fernández-Diaza
a Dermatología Hospital Universitário Lucus Augusti, Spain
b Anatomía Patológica, Hospital Universitário Lucus Augusti, Spain
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    "titulo" => "Recurrent episodes of periorbital edema in an elderly woman"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">An 81-year-old woman was referred for an edematous&#44; 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&#46; The lesions&#44; present on both cheeks&#44; initially had a papular-vesicular appearance&#44; but they subsequently increased in size and became firm and tender and evolved rapidly into edematous plaques&#46; The patient reported pruritus and fever of 38<span class="elsevierStyleHsp" style=""></span>&#176;C&#46; There was no history of insect bite&#44; trauma&#44; foreign travel&#44; or gastrointestinal problems in the previous months&#46; The patient was not taking any regular medication&#44; including topical medication&#44; at the time of onset of the lesions&#44; nor did she use cosmetic preparations or contact lenses&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">Three years earlier she had attended the infectious diseases department with a sore&#44; pruritic erythematous plaque affecting the whole left side of the face&#44; associated with fever&#46; Antimicrobial therapy with intravenous amoxicillin plus clavulanic acid had been administered and the lesion resolved within a week&#44; without scarring&#46; A diagnosis of a probable acute bacterial cellulitis had been made&#46; There was no other medical history of note and her family history was unremarkable&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">On physical examination&#44; the patient had a healthy appearance&#44; was afebrile&#44; and presented a tender&#44; infiltrated erythematous plaque that affected the periorbital region and cheek on the right side of the face &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; There were no palpable lymph nodes and no other skin lesions&#46; The total IgE concentration was 1334<span class="elsevierStyleHsp" style=""></span>kU&#47;L &#40;normal range&#44; 0&#8211;100<span class="elsevierStyleHsp" style=""></span>kU&#47;L&#41;&#46; Complete blood count&#44; biochemistry&#44; and serologic tests for antinuclear antibodies&#44; antineutrophil cytoplasmic antibodies&#44; and C1 inhibitor and C4 levels were normal or negative&#46; Chest and sinus X-rays were normal&#46; Facial computed tomography showed no ocular or intracranial alterations&#46; Ophthalmologic examination was normal in both eyes and no ocular dysfunction was detected&#46; Direct immunofluorescence for herpes viruses was negative&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The suspected clinical diagnosis was an insect bite reaction with acute cellulitis&#46; The patient was treated with intravenous cloxacillin and the skin lesions improved within a few days&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Two weeks later&#44; the patient had a recurrence&#44; with the reappearance of an erythematous&#44; edematous plaque in the right periocular area&#44; associated with bullous papules on both arms and hands&#46; These lesions were initially bright red&#44; but progressively darkened to a violaceous color&#44; giving the appearance of hematomas &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; There was no malaise or fever&#46; Laboratory studies were within normal limits&#44; including the peripheral eosinophil count&#46; The total IgE concentration was 910<span class="elsevierStyleHsp" style=""></span>kU&#47;L&#46;</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&#46; Additionally&#44; degranulated material and fragmented eosinophils were observed around collagen fibers in the dermis and were identified as flame figures &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Direct immunofluorescence was negative&#46; A diagnosis of Wells syndrome was made&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Oral steroid therapy was started at 0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;d and the patient&#39;s cutaneous signs and symptoms improved within 24<span class="elsevierStyleHsp" style=""></span>h&#44; leaving residual lesions&#46; The dose of oral prednisone was tapered over 6 days&#46; However&#44; 10 days after withdrawal of the corticosteroid treatment&#44; the patient presented recurrence in the form of edema in the left periorbital region and vesicles on forehead&#59; there were no systemic symptoms at that time&#46; A further cycle of oral steroids &#40;prednisone 30<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41; was administered&#44; leading to a notable improvement&#44; but a new flare-up occurred when the dose was reduced below 10<span class="elsevierStyleHsp" style=""></span>mg a day&#46; Oral prednisone&#44; 10<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#44; was therefore continued for a month&#44; with no adverse effects&#46; At the end of that cycle&#44; the lesions had cleared completely and the patient has subsequently remained symptom-free&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Wells syndrome is a rare dermatosis of unknown etiology and pathogenesis&#46; It is typically characterized by recurrent episodes of pruritic plaques&#44; occasionally associated with bullae&#44; which develop over 2&#8211;3 days and usually resolve spontaneously over 2&#8211;8 weeks&#46; The site and appearance of the skin lesions vary&#44; but they often affect the limbs&#46; Facial lesions are rare&#46; In our patient&#44; the periorbital region was the most severely affected area&#44; alternating from right to left sides in successive recurrences&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The most common systemic complaint is malaise&#46; Fever is present in less than a quarter of cases&#46; The manifestations of eosinophilic cellulitis are difficult to differentiate from bacterial cellulitis&#44; and misdiagnosis is common&#46; In our case&#44; the first skin lesions were associated with fever&#44; leading to a misdiagnosis of bacterial cellulitis&#46; Although there was an initial improvement with antimicrobial therapy&#44; we consider this as a coincidence and that the cutaneous lesions had probably undergone spontaneous remission&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Approximately 50&#37; of cases reported in the literature have presented peripheral blood eosinophilia during active disease&#44; but the eosinophil count remained normal in our patient&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">4&#8211;8</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion&#44; we have described a patient initially diagnosed with bacterial cellulitis&#44; but in whom skin biopsy subsequently established a diagnosis of eosinophilic cellulitis&#46; This occurred in the absence of any obvious trigger or associated skin conditions and showed a good response to steroid therapy&#46; We would like to draw attention to the atypical periorbital site of the lesions&#46;</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 &#40;<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#46;<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&#46;</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&#46;</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&#46;</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&#46;</p></span></span>"
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ISSN: 00017310
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