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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 30-year-old woman was seen for skin lesions on both legs that occurred in sporadic flares and were associated with localized itching&#46; Notably&#44; the patient&#39;s medical history included a diagnosis of Sj&#246;gren syndrome with sicca&#44; based on compatible histological findings in a salivary gland biopsy and the presence of anti-Ro&#47;SSa &#40;Sj&#246;gren-syndrome-related antigen A&#41; and anti-La&#47;SSb<span class="elsevierStyleSup">&#43;</span> &#40;Sj&#246;gren-syndrome-related antigen &#946;&#41; antinuclear antibodies&#46; The lesions consisted of nonpalpable purpura that persisted for approximately 5 days after exercise&#44; prolonged standing&#44; and stress&#44; although the patient also reported the appearance of lesions in the absence of activity&#46; In some cases the lesions were accompanied by pain and inflammation of the knees and ankles&#46; The patient reported no fever or other systemic symptoms&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">Examination revealed nonpalpable petechiae and purpuric spots&#44; which did not blanch on finger pressure&#44; distributed bilaterally and symmetrically on the legs&#44; confluent lesions on the thighs&#44; and isolated petechiae on the dorsal aspect of the feet &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; A striking finding was the presence of the Rumpel-Leede phenomenon on the insides of both legs&#44; coinciding with the seam of the pants&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In addition to the aforementioned anti-Ro&#47;SSa and anti-La&#47;SSb antinuclear antibodies&#44; laboratory analyses revealed the following&#58; elevated erythrocyte sedimentation rate &#40;ESR&#59; 76<span class="elsevierStyleHsp" style=""></span>mm&#41;&#59; rheumatoid factor&#44; 84<span class="elsevierStyleHsp" style=""></span>IU&#47;mL&#59; IgG&#44; 2680<span class="elsevierStyleHsp" style=""></span>mg&#47;dL &#40;polyclonal hypergammaglobulinemia on immunofixation&#41;&#46; The remaining tests &#40;complete blood count&#44; coagulation study&#44; basic biochemistry&#44; serology&#44; and complement analysis&#41; revealed no alterations&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Histology of a biopsy of one of the purpuric macules showed a dense&#44; lymphocytic and polymorphonuclear perivascular infiltrate with leukocytoclasia&#44; without fibrinoid necrosis&#44; in the upper papillary and reticular dermis &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; Direct immunofluorescence &#40;DIF&#41; was negative&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">What Is Your Diagnosis&#63;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">Hypergammaglobulinemic purpura of Waldenstr&#246;m&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Comment</span><p id="par0035" class="elsevierStylePara elsevierViewall">A diagnosis of hypergammaglobulinemic purpura of Waldenstr&#246;m was established based on the recurrent flares of purpura on the lower limbs&#44; the patient&#39;s clinical history &#40;Sj&#246;gren syndrome&#41;&#44; and the laboratory &#40;hypergammaglobulinemia and elevated rheumatoid factor and ESR levels&#41; and histological findings&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Hypergammaglobulinemic purpura of Waldenstr&#246;m is a rare entity that is common in middle-aged women and consists of flares of petechial lesions&#46; These lesions predominantly affect the lower limbs&#44; can coalesce to form purpuric spots &#40;as seen in our patient&#41;&#44; and rarely become palpable&#46; Lesions can be accompanied by sensations of itching&#44; burning&#44; or even pain&#46; Flares occur at highly variable intervals&#44; ranging from weeks to years&#46; In some cases the clinical picture can be accompanied by lymphadenopathy&#44; arthritis&#44; or fever&#46; Relapses are usually triggered by increased hydrostatic pressure &#40;e&#46;g&#46; due to prolonged standing&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Two forms of purpura of Waldenstr&#246;m are recognized&#58; primary&#44; which is not associated with other diseases&#59; and secondary&#44; which is more frequent&#44; and is associated with autoimmune disorders&#44; granulomatous diseases&#44; and blood dyscrasias&#46; The secondary form is most commonly associated with Sj&#246;gren syndrome&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> followed by lupus erythematosus&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">3</span></a> Raynaud phenomenon&#44; and multiple myeloma&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Key laboratory findings include polyclonal hypergammaglobulinemia&#44; due to increased levels of IgG&#44; as well as IgA and IgM&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a> Elevated ESR levels typically coincide with flares&#46; Rheumatoid factor is usually present at high titers&#46; Other potential findings include usually mild alterations in the complete blood count &#40;anemia&#44; leukopenia&#41;&#44; positive ANA titers&#44; and&#44; less frequently&#44; hypocomplementemia or cryoglobulinemia&#44; which can cause diagnostic confusion with other autoimmune diseases&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Histology reveals 2 equally frequent patterns&#58; leukocytoclastic vasculitis and lymphocytic perivascular infiltrate without leukocytoclasia&#46; The infiltrate is usually predominantly polymorphonuclear in early lesions &#40;less than 24<span class="elsevierStyleHsp" style=""></span>h after onset&#41;&#44; with hematic extravasation and fibrinoid necrosis&#44; later progressing to a mononuclear or mixed infiltrate&#46; DIF findings &#40;IgG&#44; IgA&#44; and IgM&#41; are rarely positive&#44; and knowledge of time since lesion onset is essential for interpretation of this parameter&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The differential diagnosis includes leukocytoclastic vasculitis and urticaria vasculitis&#46; The latter was considered less likely in our case&#58; the lesions were highly purpuric&#44; slightly pruritic&#44; and painless&#44; without residual hyperpigmentation&#59; hypocomplementemia was absent&#59; and DIF findings were negative&#46; Moreover&#44; the presence of hypergammaglobulinemia further supported a diagnosis of purpura of Waldenstr&#246;m&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">The benign and generally asymptomatic nature of this disease makes use of aggressive treatments unnecessary in most cases&#46; Relative rest with legs raised is usually recommended&#44; occasionally accompanied by medium-potency topical corticosteroid treatment&#46; Cases treated with hydroxychloroquine&#44; indomethacin&#44; plasmapheresis&#44; aspirin&#44; colchicine&#44; and maintenance oral corticosteroids have also been described&#44; with variable results&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of Interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case for Diagnosis
Nonpalpable Purpura in a Patient With Sjögren Syndrome
Púrpura no palpable en paciente con síndrome de Sjögren
M. Agud-Diosa,
Autor para correspondencia
agudmanuel@gmail.com

Corresponding author.
, J.L. Rodríguez-Peraltob, V. Velasco-Tamariza
a Departamento de Dermatología, Hospital Universitario 12 de Octubre, Madrid, Spain
b Departamento de Anatomía Patológica, Hospital Universitario 12 de Octubre, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 30-year-old woman was seen for skin lesions on both legs that occurred in sporadic flares and were associated with localized itching&#46; Notably&#44; the patient&#39;s medical history included a diagnosis of Sj&#246;gren syndrome with sicca&#44; based on compatible histological findings in a salivary gland biopsy and the presence of anti-Ro&#47;SSa &#40;Sj&#246;gren-syndrome-related antigen A&#41; and anti-La&#47;SSb<span class="elsevierStyleSup">&#43;</span> &#40;Sj&#246;gren-syndrome-related antigen &#946;&#41; antinuclear antibodies&#46; The lesions consisted of nonpalpable purpura that persisted for approximately 5 days after exercise&#44; prolonged standing&#44; and stress&#44; although the patient also reported the appearance of lesions in the absence of activity&#46; In some cases the lesions were accompanied by pain and inflammation of the knees and ankles&#46; The patient reported no fever or other systemic symptoms&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">Examination revealed nonpalpable petechiae and purpuric spots&#44; which did not blanch on finger pressure&#44; distributed bilaterally and symmetrically on the legs&#44; confluent lesions on the thighs&#44; and isolated petechiae on the dorsal aspect of the feet &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; A striking finding was the presence of the Rumpel-Leede phenomenon on the insides of both legs&#44; coinciding with the seam of the pants&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In addition to the aforementioned anti-Ro&#47;SSa and anti-La&#47;SSb antinuclear antibodies&#44; laboratory analyses revealed the following&#58; elevated erythrocyte sedimentation rate &#40;ESR&#59; 76<span class="elsevierStyleHsp" style=""></span>mm&#41;&#59; rheumatoid factor&#44; 84<span class="elsevierStyleHsp" style=""></span>IU&#47;mL&#59; IgG&#44; 2680<span class="elsevierStyleHsp" style=""></span>mg&#47;dL &#40;polyclonal hypergammaglobulinemia on immunofixation&#41;&#46; The remaining tests &#40;complete blood count&#44; coagulation study&#44; basic biochemistry&#44; serology&#44; and complement analysis&#41; revealed no alterations&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Histology of a biopsy of one of the purpuric macules showed a dense&#44; lymphocytic and polymorphonuclear perivascular infiltrate with leukocytoclasia&#44; without fibrinoid necrosis&#44; in the upper papillary and reticular dermis &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; Direct immunofluorescence &#40;DIF&#41; was negative&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">What Is Your Diagnosis&#63;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">Hypergammaglobulinemic purpura of Waldenstr&#246;m&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Comment</span><p id="par0035" class="elsevierStylePara elsevierViewall">A diagnosis of hypergammaglobulinemic purpura of Waldenstr&#246;m was established based on the recurrent flares of purpura on the lower limbs&#44; the patient&#39;s clinical history &#40;Sj&#246;gren syndrome&#41;&#44; and the laboratory &#40;hypergammaglobulinemia and elevated rheumatoid factor and ESR levels&#41; and histological findings&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Hypergammaglobulinemic purpura of Waldenstr&#246;m is a rare entity that is common in middle-aged women and consists of flares of petechial lesions&#46; These lesions predominantly affect the lower limbs&#44; can coalesce to form purpuric spots &#40;as seen in our patient&#41;&#44; and rarely become palpable&#46; Lesions can be accompanied by sensations of itching&#44; burning&#44; or even pain&#46; Flares occur at highly variable intervals&#44; ranging from weeks to years&#46; In some cases the clinical picture can be accompanied by lymphadenopathy&#44; arthritis&#44; or fever&#46; Relapses are usually triggered by increased hydrostatic pressure &#40;e&#46;g&#46; due to prolonged standing&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Two forms of purpura of Waldenstr&#246;m are recognized&#58; primary&#44; which is not associated with other diseases&#59; and secondary&#44; which is more frequent&#44; and is associated with autoimmune disorders&#44; granulomatous diseases&#44; and blood dyscrasias&#46; The secondary form is most commonly associated with Sj&#246;gren syndrome&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> followed by lupus erythematosus&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">3</span></a> Raynaud phenomenon&#44; and multiple myeloma&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Key laboratory findings include polyclonal hypergammaglobulinemia&#44; due to increased levels of IgG&#44; as well as IgA and IgM&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a> Elevated ESR levels typically coincide with flares&#46; Rheumatoid factor is usually present at high titers&#46; Other potential findings include usually mild alterations in the complete blood count &#40;anemia&#44; leukopenia&#41;&#44; positive ANA titers&#44; and&#44; less frequently&#44; hypocomplementemia or cryoglobulinemia&#44; which can cause diagnostic confusion with other autoimmune diseases&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Histology reveals 2 equally frequent patterns&#58; leukocytoclastic vasculitis and lymphocytic perivascular infiltrate without leukocytoclasia&#46; The infiltrate is usually predominantly polymorphonuclear in early lesions &#40;less than 24<span class="elsevierStyleHsp" style=""></span>h after onset&#41;&#44; with hematic extravasation and fibrinoid necrosis&#44; later progressing to a mononuclear or mixed infiltrate&#46; DIF findings &#40;IgG&#44; IgA&#44; and IgM&#41; are rarely positive&#44; and knowledge of time since lesion onset is essential for interpretation of this parameter&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The differential diagnosis includes leukocytoclastic vasculitis and urticaria vasculitis&#46; The latter was considered less likely in our case&#58; the lesions were highly purpuric&#44; slightly pruritic&#44; and painless&#44; without residual hyperpigmentation&#59; hypocomplementemia was absent&#59; and DIF findings were negative&#46; Moreover&#44; the presence of hypergammaglobulinemia further supported a diagnosis of purpura of Waldenstr&#246;m&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">The benign and generally asymptomatic nature of this disease makes use of aggressive treatments unnecessary in most cases&#46; Relative rest with legs raised is usually recommended&#44; occasionally accompanied by medium-potency topical corticosteroid treatment&#46; Cases treated with hydroxychloroquine&#44; indomethacin&#44; plasmapheresis&#44; aspirin&#44; colchicine&#44; and maintenance oral corticosteroids have also been described&#44; with variable results&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of Interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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