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ferritin &#40;12&#44;000<span class="elsevierStyleHsp" style=""></span>ng&#47;mL&#41; and C-reactive protein &#40;8<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41;&#46; Antistreptolysin titers&#44; antinuclear antibody&#44; rheumatoid factor&#44; antineutrophil cytoplasmic antibodies&#44; serum creatine phosphokinase&#44; and aldolase were negative or within normal limits&#46; Blood and urine cultures and other investigations for hepatitis B&#44; C and A viruses&#44; and chikungunya were all negative&#46; Chest radiograph&#44; echocardiogram&#44; ultrasonography of abdomen and pelvis&#44; and electromyogram did not reveal any abnormality&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Histological examination revealed microabscesses in the stratum corneum with necrotic keratinocytes and perivascular and periadnexal inflammatory infiltrate &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Considering her clinical and histological findings&#44; diagnosis of adult onset Still&#39;s disease &#40;AOSD&#41; was made and she was treated with oral steroid &#40;1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#41;&#46; There was significant resolution of her cutaneous lesions and other symptoms also ameliorated at the 4 weeks of follow-up&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Still&#39;s disease is an idiopathic systemic inflammatory disorder with seronegative arthritis&#46; Patients with 16 years of age or older having Sill&#39;s disease are labeled as AOSD&#44; whereas younger than 16 years are termed as Juvenile Still&#39;s disease&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Yamaguchi et al&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> have led the major diagnostic criteria to diagnose AOSD&#44; which include high spiking fevers &#8805;39<span class="elsevierStyleHsp" style=""></span>&#176;C for at least 1 week&#44; leukocytosis with neutrophilia&#44; arthralgia for more than 2 weeks&#44; and typical skin eruption&#59; whereas the minor criteria include sore throat&#44; lymphadenopathy&#44; splenomegaly&#44; hepatic dysfunction&#44; negative rheumatoid factor and antinuclear antibody&#46; AOSD is diagnosed when five or more criteria &#40;including 2 major criteria&#41; are present in the patient&#44; provided there is no evidence of infections&#44; malignant conditions&#44; and other rheumatologic diseases&#46; However Sun NZ et al&#46; have found the delayed association of malignancies &#40;breast cancer and lymphoma&#41; with AOSD&#44; especially in patients having atypical cutaneous lesions&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The classic rash of AOSD consists of an evanescent&#44; non-pruritic salmon pink&#44; morbilliform eruption on the trunk and&#47;or extremities which appears simultaneously with fever spikes&#46; The non-classic variant of the skin rash in AOSD which is characterized by pruritic papules and plaques with fine scales along with flagellate erythema located on the trunk&#44; extremities&#44; head and&#47;or neck&#46; Owing to its itchy nature&#44; the linearity of this rash over especially over extremities may represent a Koebner phenomenon&#46; Flagellate erythema have been observed in patients undergoing chemotherapy with bleomycin&#44; peplomycin&#44; docetaxel and transtuzumab and also in patients suffering from dermatomyositis&#44; systemic lupus erythematosus&#44; chikungunya fever and Parvoirus B19 infection and after the consumption of shiitake mushroom&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The histopathologic findings of AOSD are nonspecific&#44; which include mild superficial perivascular lymphocytic infiltrate with variable neutrophils&#44; whereas the histopathology of atypical rash of AOSD include dyskeratosis or parakeratosis and necrotic keratinocytes in the upper layers of the epidermis extending into the stratum corneum and a sparse superficial dermal infiltrate containing neutrophils without vasculitis&#46; In some cases dermal mucin deposits are present&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Erythema multiforme is the prototype diagnosis within the histologic differential of necrotic keratinocytes&#44; which also includes Stevens-Johnson syndrome&#44; toxic epidermal necrolysis&#44; lichen planus&#44; lupus&#44; and graft-versus-host disease&#46; However in EM the necrotic keratinocytes are scattered throughout the epidermis&#44; whereas in Still&#39;s disease they are situated in the upper epidermis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Other atypical skin lesions less commonly seen in AOSD include urticaria and urticaria-like eruptions&#44; generalized non-pruritic persistent erythema&#44; vesiculopustular eruptions&#44; a widespread peau d&#8217;orange appearance of the skin and edema of the eyelids mimicking dermatomyositis&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> Recently few atypical forms of AOSD reported include neutrophilic urticarial dermatosis&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> brown macules on oral mucosa<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> and generealised purpura&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The majorities of patients of AOSD with atypical cutaneous lesions have high levels of ferritin and they often suffer from persistent and severe disease&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Most patients respond to medium or high doses of glucocorticoids while those with severe and persistent disease need a more potent immunosuppressant drugs including methotrexate&#44; azathioprine&#44; cyclosporine A&#44; hydroxychloroquin and IL-1 receptor antagonist&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Treating physician should be aware of the atypical cutaneous features of AOSD as it is a poor prognostic marker needing prompt diagnosis and aggressive treatment measures&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">This work has not received any type of funding&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest&#46;</p></span></span>"
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Case and Research Letters
Persistent Pruritic Papules AND Plaques and Flagellate Erythema as Presenting Manifestations of an Adult onset Still's Disease
Pápulas y placas pruriginosas persistentes y eritema flagelado como manifestaciones de una enfermedad de Still del adulto
M. Pawar
Autor para correspondencia
manojpawar624@yahoo.com

Corresponding author.
, V. Zawar, S. Kumavat
Departamento de Dermatología, Dr. V. P. Facultad de Medicina, Hospital y Centro de Investigación, Nashik, India
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ferritin &#40;12&#44;000<span class="elsevierStyleHsp" style=""></span>ng&#47;mL&#41; and C-reactive protein &#40;8<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41;&#46; Antistreptolysin titers&#44; antinuclear antibody&#44; rheumatoid factor&#44; antineutrophil cytoplasmic antibodies&#44; serum creatine phosphokinase&#44; and aldolase were negative or within normal limits&#46; Blood and urine cultures and other investigations for hepatitis B&#44; C and A viruses&#44; and chikungunya were all negative&#46; Chest radiograph&#44; echocardiogram&#44; ultrasonography of abdomen and pelvis&#44; and electromyogram did not reveal any abnormality&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Histological examination revealed microabscesses in the stratum corneum with necrotic keratinocytes and perivascular and periadnexal inflammatory infiltrate &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Considering her clinical and histological findings&#44; diagnosis of adult onset Still&#39;s disease &#40;AOSD&#41; was made and she was treated with oral steroid &#40;1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#41;&#46; There was significant resolution of her cutaneous lesions and other symptoms also ameliorated at the 4 weeks of follow-up&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Still&#39;s disease is an idiopathic systemic inflammatory disorder with seronegative arthritis&#46; Patients with 16 years of age or older having Sill&#39;s disease are labeled as AOSD&#44; whereas younger than 16 years are termed as Juvenile Still&#39;s disease&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Yamaguchi et al&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> have led the major diagnostic criteria to diagnose AOSD&#44; which include high spiking fevers &#8805;39<span class="elsevierStyleHsp" style=""></span>&#176;C for at least 1 week&#44; leukocytosis with neutrophilia&#44; arthralgia for more than 2 weeks&#44; and typical skin eruption&#59; whereas the minor criteria include sore throat&#44; lymphadenopathy&#44; splenomegaly&#44; hepatic dysfunction&#44; negative rheumatoid factor and antinuclear antibody&#46; AOSD is diagnosed when five or more criteria &#40;including 2 major criteria&#41; are present in the patient&#44; provided there is no evidence of infections&#44; malignant conditions&#44; and other rheumatologic diseases&#46; However Sun NZ et al&#46; have found the delayed association of malignancies &#40;breast cancer and lymphoma&#41; with AOSD&#44; especially in patients having atypical cutaneous lesions&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The classic rash of AOSD consists of an evanescent&#44; non-pruritic salmon pink&#44; morbilliform eruption on the trunk and&#47;or extremities which appears simultaneously with fever spikes&#46; The non-classic variant of the skin rash in AOSD which is characterized by pruritic papules and plaques with fine scales along with flagellate erythema located on the trunk&#44; extremities&#44; head and&#47;or neck&#46; Owing to its itchy nature&#44; the linearity of this rash over especially over extremities may represent a Koebner phenomenon&#46; Flagellate erythema have been observed in patients undergoing chemotherapy with bleomycin&#44; peplomycin&#44; docetaxel and transtuzumab and also in patients suffering from dermatomyositis&#44; systemic lupus erythematosus&#44; chikungunya fever and Parvoirus B19 infection and after the consumption of shiitake mushroom&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The histopathologic findings of AOSD are nonspecific&#44; which include mild superficial perivascular lymphocytic infiltrate with variable neutrophils&#44; whereas the histopathology of atypical rash of AOSD include dyskeratosis or parakeratosis and necrotic keratinocytes in the upper layers of the epidermis extending into the stratum corneum and a sparse superficial dermal infiltrate containing neutrophils without vasculitis&#46; In some cases dermal mucin deposits are present&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Erythema multiforme is the prototype diagnosis within the histologic differential of necrotic keratinocytes&#44; which also includes Stevens-Johnson syndrome&#44; toxic epidermal necrolysis&#44; lichen planus&#44; lupus&#44; and graft-versus-host disease&#46; However in EM the necrotic keratinocytes are scattered throughout the epidermis&#44; whereas in Still&#39;s disease they are situated in the upper epidermis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Other atypical skin lesions less commonly seen in AOSD include urticaria and urticaria-like eruptions&#44; generalized non-pruritic persistent erythema&#44; vesiculopustular eruptions&#44; a widespread peau d&#8217;orange appearance of the skin and edema of the eyelids mimicking dermatomyositis&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> Recently few atypical forms of AOSD reported include neutrophilic urticarial dermatosis&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> brown macules on oral mucosa<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> and generealised purpura&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The majorities of patients of AOSD with atypical cutaneous lesions have high levels of ferritin and they often suffer from persistent and severe disease&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Most patients respond to medium or high doses of glucocorticoids while those with severe and persistent disease need a more potent immunosuppressant drugs including methotrexate&#44; azathioprine&#44; cyclosporine A&#44; hydroxychloroquin and IL-1 receptor antagonist&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Treating physician should be aware of the atypical cutaneous features of AOSD as it is a poor prognostic marker needing prompt diagnosis and aggressive treatment measures&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">This work has not received any type of funding&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest&#46;</p></span></span>"
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ISSN: 15782190
Idioma original: Inglés
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