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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Necrobiosis lipoidica &#40;NL&#41; is a granulomatous disease of unknown etiology that typically occurs in diabetic patients&#46; It is characterized by sclerotic plaques that most often appear on the legs&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 15-year-old girl was admitted to our hospital with facial lesions that had appeared 5 months earlier&#46; She had previously consulted with a surgeon who had suggested surgical excision of the lesions&#46; Physical examination revealed yellowish nodules and plaques with superficial telangiectasias on the upper and lower eyelids of the left eye &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The patient had been diagnosed with type 1 diabetes mellitus at age 6 years and had a history of marked insulin resistance and chronic poor blood glucose control &#40;hemoglobin A<span class="elsevierStyleInf">1c</span>&#44; 13&#37;&#41;&#46; Skin biopsy revealed an inflammatory infiltrate in the reticular dermis and hypodermis&#44; predominantly composed of histiocytes that had aggregated to form granulomas surrounded by degenerated collagen fibers&#46; Other findings included extracellular lipid deposits and no increase in stromal mucin&#46; Plasma cells and multinucleated giant cells were also observed &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; A diagnosis of NL was established on the basis of clinical and histologic findings and the patient&#39;s past history&#46; Because the site of the skin lesions made the use of topical or intralesional corticosteroids inadvisable&#44; treatment was started with twice daily applications of 0&#46;1&#37; tacrolimus ointment&#46; After 2 months of treatment with no improvement&#44; oral pentoxifylline &#40;600<span class="elsevierStyleHsp" style=""></span>mg&#47;12 h&#41; was added but was subsequently discontinued owing to gastrointestinal intolerance&#46; When the lesions had resolved only partially after 4 months of treatment&#44; topical tacrolimus was replaced with acetylsalicylic acid &#40;300<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41;&#46; After a further 10 weeks of treatment&#44; the periocular plaques had clearly improved&#44; but other plaques of NL&#44; including 1 with ulceration&#44; appeared in the pretibial region&#46; The pretibial plaques were treated with intralesional corticosteroids and hyperbaric oxygen therapy and resolved almost completely&#46; After 2 years of treatment with acetylsalicylic acid&#44; the periocular lesions had decreased in size until only a few small&#44; infiltrated&#44; slightly erythematous plaques remained&#46; Ten months after withdrawal of the treatment&#44; the plaques had not increased in size&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">NL is 3 times more common in women than in men&#44; with the age of onset usually between 30 and 40 years&#46; It is often associated with type 1 diabetes mellitus&#59; between 75&#37; and 90&#37; of patients with NL have or will develop diabetes&#44; although NL is present in only 0&#46;3&#37; to 3&#37; of diabetic patients&#46; NL has also been associated with autoimmune thyroid disease&#44; rheumatoid arthritis&#44; inflammatory bowel disease&#44; and sarcoidosis&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Although NL lesions are typically located on the anterior and lateral aspects of the legs&#44; cases affecting the trunk&#44; upper limbs&#44; face&#44; and penis have also been described&#46; NL lesions in the periocular region are extremely rare&#44; but they have been found both in isolation<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> and in association with NL lesions in other areas&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The lesion begins as an erythematous papule or plaque&#44; which extends peripherally&#44; developing a yellowish atrophic center and a raised erythematous border&#46; The main complication of NL is ulceration&#44; which occurs in 25&#37; to 33&#37; of patients&#46; Atypical lesions on the face and the edge of the scalp have been described<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> in association with Miescher granuloma&#44; actinic granuloma&#44; granuloma multiforme&#44; and necrobiotic xanthogranuloma&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">NL is histologically characterized by a normal or atrophic&#8212;and frequently ulcerated&#8212;epidermis and necrobiotic collagen with sclerosis&#46; In the dermis&#44; palisading granulomas arranged in layers parallel to the epidermis can be observed&#46; The dermal inflammatory infiltrate is composed of histiocytes&#44; multinucleated giant cells&#44; lymphocytes&#44; and plasma cells&#46; The histopathological differential diagnosis must primarily rule out granuloma annulare&#44; rheumatoid nodules&#44; and necrobiotic xanthogranuloma&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In cases with periorbital involvement it is essential to rule out the possibility of necrobiotic xanthogranuloma&#44; a form of histiocytosis associated with paraproteinemia that usually occurs in periorbital sites&#46; This disorder manifests as yellowish-red indurated nodules or plaques&#44; frequently with atrophy&#44; ulceration&#44; and telangiectasias&#46; Histologically&#44; necrobiotic xanthogranuloma is differentiated from NL by its denser infiltrate of histiocytes&#44; more pronounced inflammation and more severe degeneration of the subcutaneous cell tissue&#44; and the presence of foamy histiocytes&#44; Touton giant cells&#44; bizarre foreign-body giant cells&#44; and cholesterol clefts&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">NL is treated for cosmetic reasons or to avoid ulceration and the risk of secondary infection and deep tissue destruction&#46; Topical and intralesional corticosteroids are the drugs most widely used to treat NL&#46; Other drugs that have been used include acetylsalicylic acid&#44; ticlopidine&#44; pentoxifylline&#44; tretinoin&#44; clofazimine&#44; mycophenolate mofetil&#44; tumor necrosis factor &#40;TNF&#41; inhibitors&#44; ciclosporin&#44; thalidomide&#44; fumaric acid esters&#44; hydroxychloroquine&#44; niacinamide&#44; photodynamic therapy&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> psoralen&#8211;UV-A&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> and topical tacrolimus&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Blood glucose control and treatment for diabetes do not appear to have any beneficial effect on NL lesions&#46; Surgical excision of the affected area can ensure resolution of the lesions but may also lead to considerable cosmetic and functional sequelae in the case of periorbital lesions&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In conclusion&#44; when NL lesions occur in atypical sites&#44; a high index of suspicion is required if we are to avoid misdiagnoses and inappropriate treatment decisions&#46; Although it was only partially effective in the case of our patient&#44; topical tacrolimus appears to be effective and particularly safe for the treatment of periocular NL lesions&#46;</p></span>"
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Case and Research Letter
Periorbital Necrobiosis Lipoidica
Necrobiosis lipoidica periorbitaria
G. Pitarcha,
Autor para correspondencia
gerardpitarch@hotmail.com

Corresponding author.
, F. Ginerb
a Servicio de Dermatología, Hospital General de Castellón, Castellón, Spain
b Servicio de Anatomía Patológica, Hospital General de Castellón, Castellón, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Necrobiosis lipoidica &#40;NL&#41; is a granulomatous disease of unknown etiology that typically occurs in diabetic patients&#46; It is characterized by sclerotic plaques that most often appear on the legs&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 15-year-old girl was admitted to our hospital with facial lesions that had appeared 5 months earlier&#46; She had previously consulted with a surgeon who had suggested surgical excision of the lesions&#46; Physical examination revealed yellowish nodules and plaques with superficial telangiectasias on the upper and lower eyelids of the left eye &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The patient had been diagnosed with type 1 diabetes mellitus at age 6 years and had a history of marked insulin resistance and chronic poor blood glucose control &#40;hemoglobin A<span class="elsevierStyleInf">1c</span>&#44; 13&#37;&#41;&#46; Skin biopsy revealed an inflammatory infiltrate in the reticular dermis and hypodermis&#44; predominantly composed of histiocytes that had aggregated to form granulomas surrounded by degenerated collagen fibers&#46; Other findings included extracellular lipid deposits and no increase in stromal mucin&#46; Plasma cells and multinucleated giant cells were also observed &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; A diagnosis of NL was established on the basis of clinical and histologic findings and the patient&#39;s past history&#46; Because the site of the skin lesions made the use of topical or intralesional corticosteroids inadvisable&#44; treatment was started with twice daily applications of 0&#46;1&#37; tacrolimus ointment&#46; After 2 months of treatment with no improvement&#44; oral pentoxifylline &#40;600<span class="elsevierStyleHsp" style=""></span>mg&#47;12 h&#41; was added but was subsequently discontinued owing to gastrointestinal intolerance&#46; When the lesions had resolved only partially after 4 months of treatment&#44; topical tacrolimus was replaced with acetylsalicylic acid &#40;300<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41;&#46; After a further 10 weeks of treatment&#44; the periocular plaques had clearly improved&#44; but other plaques of NL&#44; including 1 with ulceration&#44; appeared in the pretibial region&#46; The pretibial plaques were treated with intralesional corticosteroids and hyperbaric oxygen therapy and resolved almost completely&#46; After 2 years of treatment with acetylsalicylic acid&#44; the periocular lesions had decreased in size until only a few small&#44; infiltrated&#44; slightly erythematous plaques remained&#46; Ten months after withdrawal of the treatment&#44; the plaques had not increased in size&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">NL is 3 times more common in women than in men&#44; with the age of onset usually between 30 and 40 years&#46; It is often associated with type 1 diabetes mellitus&#59; between 75&#37; and 90&#37; of patients with NL have or will develop diabetes&#44; although NL is present in only 0&#46;3&#37; to 3&#37; of diabetic patients&#46; NL has also been associated with autoimmune thyroid disease&#44; rheumatoid arthritis&#44; inflammatory bowel disease&#44; and sarcoidosis&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Although NL lesions are typically located on the anterior and lateral aspects of the legs&#44; cases affecting the trunk&#44; upper limbs&#44; face&#44; and penis have also been described&#46; NL lesions in the periocular region are extremely rare&#44; but they have been found both in isolation<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> and in association with NL lesions in other areas&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The lesion begins as an erythematous papule or plaque&#44; which extends peripherally&#44; developing a yellowish atrophic center and a raised erythematous border&#46; The main complication of NL is ulceration&#44; which occurs in 25&#37; to 33&#37; of patients&#46; Atypical lesions on the face and the edge of the scalp have been described<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> in association with Miescher granuloma&#44; actinic granuloma&#44; granuloma multiforme&#44; and necrobiotic xanthogranuloma&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">NL is histologically characterized by a normal or atrophic&#8212;and frequently ulcerated&#8212;epidermis and necrobiotic collagen with sclerosis&#46; In the dermis&#44; palisading granulomas arranged in layers parallel to the epidermis can be observed&#46; The dermal inflammatory infiltrate is composed of histiocytes&#44; multinucleated giant cells&#44; lymphocytes&#44; and plasma cells&#46; The histopathological differential diagnosis must primarily rule out granuloma annulare&#44; rheumatoid nodules&#44; and necrobiotic xanthogranuloma&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In cases with periorbital involvement it is essential to rule out the possibility of necrobiotic xanthogranuloma&#44; a form of histiocytosis associated with paraproteinemia that usually occurs in periorbital sites&#46; This disorder manifests as yellowish-red indurated nodules or plaques&#44; frequently with atrophy&#44; ulceration&#44; and telangiectasias&#46; Histologically&#44; necrobiotic xanthogranuloma is differentiated from NL by its denser infiltrate of histiocytes&#44; more pronounced inflammation and more severe degeneration of the subcutaneous cell tissue&#44; and the presence of foamy histiocytes&#44; Touton giant cells&#44; bizarre foreign-body giant cells&#44; and cholesterol clefts&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">NL is treated for cosmetic reasons or to avoid ulceration and the risk of secondary infection and deep tissue destruction&#46; Topical and intralesional corticosteroids are the drugs most widely used to treat NL&#46; Other drugs that have been used include acetylsalicylic acid&#44; ticlopidine&#44; pentoxifylline&#44; tretinoin&#44; clofazimine&#44; mycophenolate mofetil&#44; tumor necrosis factor &#40;TNF&#41; inhibitors&#44; ciclosporin&#44; thalidomide&#44; fumaric acid esters&#44; hydroxychloroquine&#44; niacinamide&#44; photodynamic therapy&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> psoralen&#8211;UV-A&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> and topical tacrolimus&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Blood glucose control and treatment for diabetes do not appear to have any beneficial effect on NL lesions&#46; Surgical excision of the affected area can ensure resolution of the lesions but may also lead to considerable cosmetic and functional sequelae in the case of periorbital lesions&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In conclusion&#44; when NL lesions occur in atypical sites&#44; a high index of suspicion is required if we are to avoid misdiagnoses and inappropriate treatment decisions&#46; Although it was only partially effective in the case of our patient&#44; topical tacrolimus appears to be effective and particularly safe for the treatment of periocular NL lesions&#46;</p></span>"
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Información del artículo
ISSN: 15782190
Idioma original: Inglés
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