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pain&#44; and complete hemiplegia of the lower right limb&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed excoriated&#44; erythematous&#44; purpuric papules and plaques&#44; including some with central ulceration &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; No other associated lesions were observed&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">Histologic examination revealed a cutaneous ulcer with necrotizing vasculitis and a mixed neutrophilic and eosinophilic infiltrate in perivascular and interstitial areas &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; Cultures&#44; Gram stain&#44; and direct immunofluorescence were negative&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Additional Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Blood tests revealed eosinophilia &#40;19&#160;620 eosinophils&#47;&#956;l&#59; 72&#37;&#41; and perinuclear antineutrophil cytoplasmic antibodies &#40;pANCA&#41; &#40;34<span class="elsevierStyleHsp" style=""></span>IU&#47;mL&#59; &#60;<span class="elsevierStyleHsp" style=""></span>5 IU&#47;mL is normal&#41;&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Electromyography revealed mononeuritis multiplex in the right leg&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Chest radiography revealed minimal bilateral pleural effusion&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">What Is Your Diagnosis&#63;</span></p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Diagnosis</span><p id="par0040" class="elsevierStylePara elsevierViewall">Eosinophilic granulomatosis with polyangiitis &#40;EGPA&#41;&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Course and Treatment</span><p id="par0045" class="elsevierStylePara elsevierViewall">Treatment was started with intravenous methylprednisolone at a dosage of 1<span class="elsevierStyleHsp" style=""></span>g daily for 5 days&#44; followed by oral prednisone at a dosage of 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;d&#46; Adequate response was achieved&#44; except in the neurologic manifestations&#46; The patient was then removed from the clinical trial and treatment was started with rituximab&#46; Significant improvement was observed at subsequent follow-up visits&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Comment</span><p id="par0050" class="elsevierStylePara elsevierViewall">EGPA&#44; previously known as Churg-Strauss syndrome&#44; is a multisystem disease that occurs with equal frequency in both sexes and is characterized by allergic rhinitis&#44; asthma&#44; and severe eosinophilia&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> It is classified in the group of antineutrophil cytoplasmic antibody &#40;ANCA&#41;-associated vasculitides affecting small and medium-sized vessels and is the least common of the 3 entities included in this group&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Given the presence of ANCA&#44; the alteration of type 1 and type 2 T-helper cells&#44; the decrease in regulatory T-cells&#44; and eosinophilia caused by both increased synthesis and decreased apoptosis&#44; it has been suggested that EGPA may have an immunologic pathogenesis&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2&#44;3</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Several authors have reported EGPA in systemic-corticosteroid-dependent asthmatic patients following a reduction in corticosteroid dosage and the start of treatment with leukotriene modifiers and&#47;or omalizumab&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">4&#44;5</span></a> These authors concluded that the onset of the disease was not related to the start of treatment with new drugs but to the reduction in corticosteroid dosage&#44; and that therefore there was no causal relationship but rather an unmasking of underdiagnosed EGPA&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">EGPA develops in 3 phases&#58; a prodromal phase&#44; characterized by underlying atopy and late-onset asthma that is difficult to control&#59; an eosinophilic phase&#44; characterized by prominent blood eosinophilia&#59; and finally a vasculitic phase&#44; characterized by the development of necrotizing vasculitis of small and medium-sized vessels&#44; an eosinophilic infiltrate&#44; and in many cases vascular and extravascular granulomatosis&#46; The vasculitic phase is characterized by constitutional symptoms and multisystemic manifestations such as mononeuritis multiplex&#44; microhematuria&#44; and cardiac failure&#44; which is the most common cause of death&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Cutaneous manifestations&#8212;typical of the vasculitic phase&#8212;tend to include subcutaneous nodules&#44; palpable purpura&#44; and hemorrhagic maculopapular lesions&#46; These lesions occur most frequently on the lower limbs&#59; however&#44; lesions on the scalp are distinctive&#44; although not pathognomonic&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Between 40&#37; and 60&#37; of patients with EGPA are positive for ANCA&#44; typically pANCA&#44; although the presence of these antibodies does not predict activity and should not be taken into account in follow-up&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">There is no specific diagnostic test for EGPA&#59; diagnosis relies on preestablished criteria and evidence of vasculitis in the affected organ&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">Treatment involves systemic corticosteroids&#44; sometimes in association with cyclophosphamide if cardiac&#44; renal&#44; or central nervous system involvement is present&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">6&#44;7</span></a> Rituximab&#44; mepolizumab&#44; and omalizumab have been used in refractory cases&#44; with disparate results&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">Differential diagnosis should include other entities characterized by angiolymphoid hyperplasia with eosinophilia as well as hypereosinophilic syndrome&#44; as these are typically not associated with asthma or histopathologic findings indicative of vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2&#44;6</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">In conclusion&#44; we postulate that the reduction in corticosteroid dosage was the factor that triggered systemic manifestations in our patient&#44; since these drugs likely masked the EGPA&#44; which had not yet entered the vasculitic phase&#46; We highlight the characteristic presentation of vasculitic lesions on the scalp&#46; In patients with asthma&#44; severe eosinophilia&#44; systemic manifestations&#44; and skin lesions&#44; early histologic diagnosis is essential in order to begin appropriate treatment&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of Interest</span><p id="par0100" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case for Diagnosis
Vasculitis With Lesions on the Scalp as the Only Cutaneous Manifestation
Vasculitis con lesiones en cuero cabelludo como única manifestación cutánea
F.C. Benavente Villegasa,
Corresponding author
felipecbv@gmail.com

Corresponding author.
, J.M. Sánchez Motillaa, N. Rausell Fontestadb
a Servicio de Dermatología, Hospital Universitario Doctor Peset, Valencia, Spain
b Servicio de Anatomía Patológica, Hospital Universitario Doctor Peset, Valencia, Spain
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pain&#44; and complete hemiplegia of the lower right limb&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed excoriated&#44; erythematous&#44; purpuric papules and plaques&#44; including some with central ulceration &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; No other associated lesions were observed&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">Histologic examination revealed a cutaneous ulcer with necrotizing vasculitis and a mixed neutrophilic and eosinophilic infiltrate in perivascular and interstitial areas &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; Cultures&#44; Gram stain&#44; and direct immunofluorescence were negative&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Additional Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Blood tests revealed eosinophilia &#40;19&#160;620 eosinophils&#47;&#956;l&#59; 72&#37;&#41; and perinuclear antineutrophil cytoplasmic antibodies &#40;pANCA&#41; &#40;34<span class="elsevierStyleHsp" style=""></span>IU&#47;mL&#59; &#60;<span class="elsevierStyleHsp" style=""></span>5 IU&#47;mL is normal&#41;&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Electromyography revealed mononeuritis multiplex in the right leg&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Chest radiography revealed minimal bilateral pleural effusion&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">What Is Your Diagnosis&#63;</span></p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Diagnosis</span><p id="par0040" class="elsevierStylePara elsevierViewall">Eosinophilic granulomatosis with polyangiitis &#40;EGPA&#41;&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Course and Treatment</span><p id="par0045" class="elsevierStylePara elsevierViewall">Treatment was started with intravenous methylprednisolone at a dosage of 1<span class="elsevierStyleHsp" style=""></span>g daily for 5 days&#44; followed by oral prednisone at a dosage of 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;d&#46; Adequate response was achieved&#44; except in the neurologic manifestations&#46; The patient was then removed from the clinical trial and treatment was started with rituximab&#46; Significant improvement was observed at subsequent follow-up visits&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Comment</span><p id="par0050" class="elsevierStylePara elsevierViewall">EGPA&#44; previously known as Churg-Strauss syndrome&#44; is a multisystem disease that occurs with equal frequency in both sexes and is characterized by allergic rhinitis&#44; asthma&#44; and severe eosinophilia&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> It is classified in the group of antineutrophil cytoplasmic antibody &#40;ANCA&#41;-associated vasculitides affecting small and medium-sized vessels and is the least common of the 3 entities included in this group&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Given the presence of ANCA&#44; the alteration of type 1 and type 2 T-helper cells&#44; the decrease in regulatory T-cells&#44; and eosinophilia caused by both increased synthesis and decreased apoptosis&#44; it has been suggested that EGPA may have an immunologic pathogenesis&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2&#44;3</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Several authors have reported EGPA in systemic-corticosteroid-dependent asthmatic patients following a reduction in corticosteroid dosage and the start of treatment with leukotriene modifiers and&#47;or omalizumab&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">4&#44;5</span></a> These authors concluded that the onset of the disease was not related to the start of treatment with new drugs but to the reduction in corticosteroid dosage&#44; and that therefore there was no causal relationship but rather an unmasking of underdiagnosed EGPA&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">EGPA develops in 3 phases&#58; a prodromal phase&#44; characterized by underlying atopy and late-onset asthma that is difficult to control&#59; an eosinophilic phase&#44; characterized by prominent blood eosinophilia&#59; and finally a vasculitic phase&#44; characterized by the development of necrotizing vasculitis of small and medium-sized vessels&#44; an eosinophilic infiltrate&#44; and in many cases vascular and extravascular granulomatosis&#46; The vasculitic phase is characterized by constitutional symptoms and multisystemic manifestations such as mononeuritis multiplex&#44; microhematuria&#44; and cardiac failure&#44; which is the most common cause of death&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Cutaneous manifestations&#8212;typical of the vasculitic phase&#8212;tend to include subcutaneous nodules&#44; palpable purpura&#44; and hemorrhagic maculopapular lesions&#46; These lesions occur most frequently on the lower limbs&#59; however&#44; lesions on the scalp are distinctive&#44; although not pathognomonic&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Between 40&#37; and 60&#37; of patients with EGPA are positive for ANCA&#44; typically pANCA&#44; although the presence of these antibodies does not predict activity and should not be taken into account in follow-up&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">There is no specific diagnostic test for EGPA&#59; diagnosis relies on preestablished criteria and evidence of vasculitis in the affected organ&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">Treatment involves systemic corticosteroids&#44; sometimes in association with cyclophosphamide if cardiac&#44; renal&#44; or central nervous system involvement is present&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">6&#44;7</span></a> Rituximab&#44; mepolizumab&#44; and omalizumab have been used in refractory cases&#44; with disparate results&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">Differential diagnosis should include other entities characterized by angiolymphoid hyperplasia with eosinophilia as well as hypereosinophilic syndrome&#44; as these are typically not associated with asthma or histopathologic findings indicative of vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2&#44;6</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">In conclusion&#44; we postulate that the reduction in corticosteroid dosage was the factor that triggered systemic manifestations in our patient&#44; since these drugs likely masked the EGPA&#44; which had not yet entered the vasculitic phase&#46; We highlight the characteristic presentation of vasculitic lesions on the scalp&#46; In patients with asthma&#44; severe eosinophilia&#44; systemic manifestations&#44; and skin lesions&#44; early histologic diagnosis is essential in order to begin appropriate treatment&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of Interest</span><p id="par0100" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Article information
ISSN: 15782190
Original language: English
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