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perilesional eczema&#44; and an ulcer measuring 3<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>2<span class="elsevierStyleHsp" style=""></span>cm with irregular borders&#44; a fibrinous base&#44; and mild signs of infection &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; with no dysmetria of the lower limbs&#46; Complete blood count&#44; biochemistry&#44; and serologic tests were negative or normal&#46; Given the presence of atypical skin lesions&#44; a complete Doppler ultrasound examination was performed on both limbs and a skin biopsy was carried out&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histologic examination revealed the proliferation of capillaries in the deep dermis and papillary dermis&#44; fibrosis&#44; extravasated red blood cells&#44; hemosiderin deposits&#44; and tortuous vessels consistent with SBS&#46; Doppler ultrasound confirmed the presence of underlying arteriovenous fistulas of the anterior and posterior tibial arteries with arterialized venous flow distal to the fistulas &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Conservative treatment with 10 days of targeted antibiotic therapy was initiated after microbiologic culture detected the presence of <span class="elsevierStyleItalic">Staphylococcus aureus</span>&#46; Compression therapy was also initiated with 2-in-1 compression stockings with a lateral zipper&#44; as well as local wound care with a lipido-colloid dressing&#46; At 3 months&#44; the ulcer had healed and the pain had disappeared&#46; The patient has continued to use compression therapy and remains asymptomatic after 1 year &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Angiodermatitis is a group of angioproliferative diseases that manifest as skin lesions resembling Kaposi sarcoma&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;3</span></a> These entities consist of benign hyperplasia of preexisting vascular structures and can be associated with AVMs&#44; as in the case of SBS&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">SBS is a rare benign angioproliferative entity with histologic similarities to Kaposi sarcoma&#46; It was first described by Earhart et al&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> in 1974 and fewer than 20 cases have been reported&#46; SBS generally affects young men starting in the second decade of life<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> and is characterized by progressive brown or violaceous maculopapular skin lesions and underlying AVMs&#46; The lesions appear unilaterally on the lower limbs&#8212;on the dorsum of the foot&#44; on the ankle&#44; and on the calf&#8212;and are accompanied by edema&#44; local temperature increase&#44; soft-tissue hypertrophy&#44; and varicose veins as a consequence of venous stasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;6</span></a> Complications can include ulcers and verrucous lesions&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Differential diagnosis should include acroangiodermatitis of Mali&#44; which typically appears in elderly patients as a consequence of chronic venous insufficiency and develops bilaterally&#44; as well as Kaposi sarcoma&#44; lichen planus&#44; vasculitis&#44; lymphoproliferative diseases characterized by congenital arteriovenous fistulas such as Klippel-Trenaunay-Weber syndrome&#44; and iatrogenic diseases in patients who have hepatitis C or are undergoing dialysis&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4&#44;7&#44;8</span></a> Various theories have been proposed regarding the etiology and pathogenesis of SBS&#59; an increase in venous pressure resulting from AVM or ischemia caused by arteriovenous steal can stimulate the proliferation of endothelial cells&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Suspicion of SBS is purely clinical &#40;atypical venous lesions&#44; soft-tissue hypertrophy&#44; palpation of thrill&#44; or auscultation of bruit&#41; and diagnosis is confirmed by histology&#59; it is therefore important that dermatologists be familiar with this rare disease&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> Histologically&#44; SBS is characterized by the proliferation of endothelial cells&#44; angiogenesis with a lobular pattern in the deep dermis and papillary dermis&#44; and extravasated red blood cells&#44; without fusiform neoplastic cells or proliferation independent of normal structures &#40;features typical of Kaposi sarcoma&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;7</span></a> Doppler ultrasound can detect the presence of arteriovenous communications&#44;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;3</span></a> although arteriography remains the gold standard<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;7</span></a> for diagnosis and treatment&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;5</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Treatment of SBS remains controversial&#46; Most experts recommend conservative treatment<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> and preventive treatment with compression stockings and limb elevation if edema is present&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Symptomatic treatment of local complications is crucial&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> whereas specific treatment is rarely possible due to the presence of various distal communications&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#44;8</span></a> Surgery is indicated in patients with functional impotence&#44; refractory pain&#44; recurrent infection&#44; bleeding&#44; or cardiac decompensation&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5&#44;8</span></a> Amputation of the limb is sometimes the only option&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Selective embolization with different particles&#44; intravenous laser&#44; radiofrequency ablation&#44; and ultrasound-guided sclerotherapy can be alternatives in complex and symptomatic cases&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> A recent case report described the use of the glycosaminoglycan Cacipliq20 &#40;heparin sulfate analogue&#41; in a patient with SBS&#44; with complete resolution of the ulcer&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; we have described a case of a rare syndrome of angiodermatitis that manifested as a chronic painful ulcer and skin lesions resembling Kaposi sarcoma&#44; arising from an underlying arteriovenous communication&#46; We have described a new vision for diagnosis using Doppler ultrasound and review the handful of studies on controversial therapeutic measures for this disease that have been published to date&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case and Research Letters
Stewart-Bluefarb Syndrome: Case Report and Review of the Literature
Síndrome de Stewart-Bluefarb: caso clínico y revisión de la literatura
V.E. García Blanco
Corresponding author
victoriagarciablanco@outlook.es

Corresponding author.
, E. Doiz Artázcoz, M.C. Galera Martínez, M. Rodríguez Piñero
Servicio de Angiología y Cirugía Vascular, Hospital Universitario Puerta del Mar, Cádiz, Spain
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perilesional eczema&#44; and an ulcer measuring 3<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>2<span class="elsevierStyleHsp" style=""></span>cm with irregular borders&#44; a fibrinous base&#44; and mild signs of infection &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; with no dysmetria of the lower limbs&#46; Complete blood count&#44; biochemistry&#44; and serologic tests were negative or normal&#46; Given the presence of atypical skin lesions&#44; a complete Doppler ultrasound examination was performed on both limbs and a skin biopsy was carried out&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histologic examination revealed the proliferation of capillaries in the deep dermis and papillary dermis&#44; fibrosis&#44; extravasated red blood cells&#44; hemosiderin deposits&#44; and tortuous vessels consistent with SBS&#46; Doppler ultrasound confirmed the presence of underlying arteriovenous fistulas of the anterior and posterior tibial arteries with arterialized venous flow distal to the fistulas &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Conservative treatment with 10 days of targeted antibiotic therapy was initiated after microbiologic culture detected the presence of <span class="elsevierStyleItalic">Staphylococcus aureus</span>&#46; Compression therapy was also initiated with 2-in-1 compression stockings with a lateral zipper&#44; as well as local wound care with a lipido-colloid dressing&#46; At 3 months&#44; the ulcer had healed and the pain had disappeared&#46; The patient has continued to use compression therapy and remains asymptomatic after 1 year &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Angiodermatitis is a group of angioproliferative diseases that manifest as skin lesions resembling Kaposi sarcoma&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;3</span></a> These entities consist of benign hyperplasia of preexisting vascular structures and can be associated with AVMs&#44; as in the case of SBS&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">SBS is a rare benign angioproliferative entity with histologic similarities to Kaposi sarcoma&#46; It was first described by Earhart et al&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> in 1974 and fewer than 20 cases have been reported&#46; SBS generally affects young men starting in the second decade of life<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> and is characterized by progressive brown or violaceous maculopapular skin lesions and underlying AVMs&#46; The lesions appear unilaterally on the lower limbs&#8212;on the dorsum of the foot&#44; on the ankle&#44; and on the calf&#8212;and are accompanied by edema&#44; local temperature increase&#44; soft-tissue hypertrophy&#44; and varicose veins as a consequence of venous stasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;6</span></a> Complications can include ulcers and verrucous lesions&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Differential diagnosis should include acroangiodermatitis of Mali&#44; which typically appears in elderly patients as a consequence of chronic venous insufficiency and develops bilaterally&#44; as well as Kaposi sarcoma&#44; lichen planus&#44; vasculitis&#44; lymphoproliferative diseases characterized by congenital arteriovenous fistulas such as Klippel-Trenaunay-Weber syndrome&#44; and iatrogenic diseases in patients who have hepatitis C or are undergoing dialysis&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4&#44;7&#44;8</span></a> Various theories have been proposed regarding the etiology and pathogenesis of SBS&#59; an increase in venous pressure resulting from AVM or ischemia caused by arteriovenous steal can stimulate the proliferation of endothelial cells&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Suspicion of SBS is purely clinical &#40;atypical venous lesions&#44; soft-tissue hypertrophy&#44; palpation of thrill&#44; or auscultation of bruit&#41; and diagnosis is confirmed by histology&#59; it is therefore important that dermatologists be familiar with this rare disease&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> Histologically&#44; SBS is characterized by the proliferation of endothelial cells&#44; angiogenesis with a lobular pattern in the deep dermis and papillary dermis&#44; and extravasated red blood cells&#44; without fusiform neoplastic cells or proliferation independent of normal structures &#40;features typical of Kaposi sarcoma&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;7</span></a> Doppler ultrasound can detect the presence of arteriovenous communications&#44;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;3</span></a> although arteriography remains the gold standard<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;7</span></a> for diagnosis and treatment&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;5</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Treatment of SBS remains controversial&#46; Most experts recommend conservative treatment<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> and preventive treatment with compression stockings and limb elevation if edema is present&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Symptomatic treatment of local complications is crucial&#44;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> whereas specific treatment is rarely possible due to the presence of various distal communications&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#44;8</span></a> Surgery is indicated in patients with functional impotence&#44; refractory pain&#44; recurrent infection&#44; bleeding&#44; or cardiac decompensation&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5&#44;8</span></a> Amputation of the limb is sometimes the only option&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Selective embolization with different particles&#44; intravenous laser&#44; radiofrequency ablation&#44; and ultrasound-guided sclerotherapy can be alternatives in complex and symptomatic cases&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> A recent case report described the use of the glycosaminoglycan Cacipliq20 &#40;heparin sulfate analogue&#41; in a patient with SBS&#44; with complete resolution of the ulcer&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; we have described a case of a rare syndrome of angiodermatitis that manifested as a chronic painful ulcer and skin lesions resembling Kaposi sarcoma&#44; arising from an underlying arteriovenous communication&#46; We have described a new vision for diagnosis using Doppler ultrasound and review the handful of studies on controversial therapeutic measures for this disease that have been published to date&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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