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1</a>A&#41;&#46; Ultrasound showed a thickened&#44; hyperechoic epidermis with hypoechoic vascular channels in the dermis and subcutaneous tissue and no color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Patient 2</span> was a 25-year-old man with a VMM on the back of his right leg that had been present since birth &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The patient had experienced recurring episodes of ulceration&#44; pain&#44; and bleeding over the previous 4 years&#46; Cutaneous ultrasound showed a thickened&#44; hyperechoic epidermis with vascular channels in the superficial and deep dermis&#46; Again&#44; there was no color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Patient 3</span> was a 12-year-old boy with a circumferential VVM in the distal region of his left leg that had been present since birth &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; In recent years&#44; the lesion had gradually gained in volume and become increasingly hyperkeratotic&#59; it had also shown ulceration and bleeding&#46; Cutaneous ultrasound showed a thickened&#44; hyperechoic epidermis&#44; decreased echogenicity in the dermis&#44; a loss of definition between the dermis and hypodermis&#44; hypodermal thickening and heterogeneity&#44; and no color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#41;&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">VVM is uncommon and is included in the group of vascular malformations in the 2018 ISSVA classification&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It has been linked to a somatic mutation in the <span class="elsevierStyleItalic">MAPK3</span> gene&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">VVM consists of a proliferation of venous blood vessels in the dermis and subcutaneous tissue&#44; with sparing of the fascia and muscle tissue&#46; It is congenital or develops in early childhood and mainly affects the lower extremities&#46; Lesions are typically unilateral&#46; Complications include bleeding&#44; pain&#44; and ulceration&#44; especially in larger&#44; more hyperkeratotic lesions&#46; VVM is not associated with tissue hypertrophy or other developmental abnormalities&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Diagnosis is usually established by correlating clinical and pathologic findings&#44; although characteristic clinical findings typically help guide diagnosis&#46; Histology shows epidermal acanthosis and papillomatosis&#44; with compact hyperkeratosis and dilated venous vessels in the papillary and reticular dermis and subcutaneous tissue&#46; Endothelial cells are positive for <span class="elsevierStyleItalic">GLUT-1</span> and <span class="elsevierStyleItalic">WT-1</span> and negative for D2-40 on immunostaining&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Cutaneous ultrasound is a useful alternative to invasive diagnostic tests in the setting of vascular lesions&#46; The ultrasound characterization of VVM is not well established&#44; but based on our findings&#44; we propose a description that could help diagnose this entity&#46; Ultrasound features of VVM include a thickened&#44; hyperechoic epidermis&#44; a loss of definition between the dermis and epidermis&#44; hyperechoic channels in the dermis and subcutaneous tissue&#44; and absence of a color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Angiokeratoma circumscriptum is the main entity to be considered in the differential diagnosis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>A&#41;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a>&#46; Its histologic features include hyperkeratotic acanthosis and dilated capillaries in the papillary dermis&#44; with no involvement of the deep dermis or subcutaneous tissue&#46; Contrary to VVM&#44; angiokeratoma circumscriptum shows negative endothelial expression of GLUT-1 and WT-1 and positive expression of D2-40 on immunostaining&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Given the differences in the depth of involvement&#44; ultrasound could help distinguish between the 2 entities without the need for invasive tests&#46; Ultrasound features of angiokeratoma include epidermal thickening and hyperechogenicity and a hypoechoic lesion limited to the papillary dermis&#46; As with VVM&#44; there is no Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>B&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">The treatment of VVM is largely surgical&#44; but surgery can sometimes be complex&#46; No treatment is an option in the absence of cosmetic concerns or symptoms&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Pulsed dye&#44; multiplex&#44; and carbon dioxide laser therapy have been used to treat complications such as ulceration and bleeding and to achieve better cosmetic outcomes&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> It should be noted that laser therapy does not resolve deep lesions and is associated with a risk of recurrence&#46; Good results with topical and oral sirolimus have been described&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Ultrasound could also be useful for monitoring the treatment of VVM as it identifies deeper areas of involvement&#44; which can be treated surgically&#44; and more superficial areas&#44; which can be treated with laser therapy or topical sirolimus&#46; A combination of approaches may improve outcomes&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">In conclusion&#44; cutaneous ultrasound appears to be a useful tool for diagnosing VVM and differentiating it from angiokeratoma&#46; It may also be of value for planning and monitoring treatment&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">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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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Amat-Samaranch V&#44; Morales-M&#250;nera C&#44; Puig L&#44; Ro&#233; E&#46; Utilidad de la ecograf&#237;a cut&#225;nea para el diagn&#243;stico de la malformaci&#243;n venosa verrucosa&#46; Actas Dermosifiliogr&#46; 2021&#59;112&#58;958&#8211;961&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical images of verrucous venous malformations&#46; A&#44; Patient 1&#46; Verrucous erythematous violaceous plaque on the left ankle&#46; B&#44; Patient 2&#46; Erythematous&#44; violaceous&#44; hyperkeratotic plaques with satellite lesions and hemorrhagic superficial vesicles on the back of the right leg&#46; C&#44; Patient 3&#46; Erythematous&#44; violaceous plaques with a verrucous surface comprising hyperkeratotic areas and crusts involving the circumference of the distal region of the left leg&#46;</p>"
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Case and Research Letters
Diagnostic Value of Cutaneous Ultrasound in Verrucous Venous Malformation
Utilidad de la ecografía cutánea para el diagnóstico de la malformación venosa verrucosa
V. Amat-Samaranch
Corresponding author
vamat@santpau.cat

Corresponding author.
, C. Morales-Múnera, L. Puig, E. Roé
Servicio de Dermatología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Verrucous venous malformation &#40;VVM&#41; is a rare vascular lesion currently classified as a vascular malformation by the International Society for the Study of Vascular Anomalies &#40;ISSVA&#41;&#46; Diagnosis is based on the integration of clinical and pathologic findings&#46; In this article&#44; we describe the ultrasound features of 3 VVMs and propose ultrasound as a useful&#44; noninvasive diagnostic and follow-up tool that can also be used to guide and optimize treatment&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We selected 3 patients with histologically confirmed VVM from the database in our department&#46; Their lesions were assessed and compared using 22-MHz cutaneous ultrasound&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Patient 1</span> was a 10-year-old boy with a VVM on his left ankle that had been present since birth &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; Ultrasound showed a thickened&#44; hyperechoic epidermis with hypoechoic vascular channels in the dermis and subcutaneous tissue and no color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Patient 2</span> was a 25-year-old man with a VMM on the back of his right leg that had been present since birth &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The patient had experienced recurring episodes of ulceration&#44; pain&#44; and bleeding over the previous 4 years&#46; Cutaneous ultrasound showed a thickened&#44; hyperechoic epidermis with vascular channels in the superficial and deep dermis&#46; Again&#44; there was no color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Patient 3</span> was a 12-year-old boy with a circumferential VVM in the distal region of his left leg that had been present since birth &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; In recent years&#44; the lesion had gradually gained in volume and become increasingly hyperkeratotic&#59; it had also shown ulceration and bleeding&#46; Cutaneous ultrasound showed a thickened&#44; hyperechoic epidermis&#44; decreased echogenicity in the dermis&#44; a loss of definition between the dermis and hypodermis&#44; hypodermal thickening and heterogeneity&#44; and no color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C&#41;&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">VVM is uncommon and is included in the group of vascular malformations in the 2018 ISSVA classification&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It has been linked to a somatic mutation in the <span class="elsevierStyleItalic">MAPK3</span> gene&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">VVM consists of a proliferation of venous blood vessels in the dermis and subcutaneous tissue&#44; with sparing of the fascia and muscle tissue&#46; It is congenital or develops in early childhood and mainly affects the lower extremities&#46; Lesions are typically unilateral&#46; Complications include bleeding&#44; pain&#44; and ulceration&#44; especially in larger&#44; more hyperkeratotic lesions&#46; VVM is not associated with tissue hypertrophy or other developmental abnormalities&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Diagnosis is usually established by correlating clinical and pathologic findings&#44; although characteristic clinical findings typically help guide diagnosis&#46; Histology shows epidermal acanthosis and papillomatosis&#44; with compact hyperkeratosis and dilated venous vessels in the papillary and reticular dermis and subcutaneous tissue&#46; Endothelial cells are positive for <span class="elsevierStyleItalic">GLUT-1</span> and <span class="elsevierStyleItalic">WT-1</span> and negative for D2-40 on immunostaining&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Cutaneous ultrasound is a useful alternative to invasive diagnostic tests in the setting of vascular lesions&#46; The ultrasound characterization of VVM is not well established&#44; but based on our findings&#44; we propose a description that could help diagnose this entity&#46; Ultrasound features of VVM include a thickened&#44; hyperechoic epidermis&#44; a loss of definition between the dermis and epidermis&#44; hyperechoic channels in the dermis and subcutaneous tissue&#44; and absence of a color Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Angiokeratoma circumscriptum is the main entity to be considered in the differential diagnosis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>A&#41;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a>&#46; Its histologic features include hyperkeratotic acanthosis and dilated capillaries in the papillary dermis&#44; with no involvement of the deep dermis or subcutaneous tissue&#46; Contrary to VVM&#44; angiokeratoma circumscriptum shows negative endothelial expression of GLUT-1 and WT-1 and positive expression of D2-40 on immunostaining&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Given the differences in the depth of involvement&#44; ultrasound could help distinguish between the 2 entities without the need for invasive tests&#46; Ultrasound features of angiokeratoma include epidermal thickening and hyperechogenicity and a hypoechoic lesion limited to the papillary dermis&#46; As with VVM&#44; there is no Doppler signal &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>B&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">The treatment of VVM is largely surgical&#44; but surgery can sometimes be complex&#46; No treatment is an option in the absence of cosmetic concerns or symptoms&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Pulsed dye&#44; multiplex&#44; and carbon dioxide laser therapy have been used to treat complications such as ulceration and bleeding and to achieve better cosmetic outcomes&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> It should be noted that laser therapy does not resolve deep lesions and is associated with a risk of recurrence&#46; Good results with topical and oral sirolimus have been described&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Ultrasound could also be useful for monitoring the treatment of VVM as it identifies deeper areas of involvement&#44; which can be treated surgically&#44; and more superficial areas&#44; which can be treated with laser therapy or topical sirolimus&#46; A combination of approaches may improve outcomes&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">In conclusion&#44; cutaneous ultrasound appears to be a useful tool for diagnosing VVM and differentiating it from angiokeratoma&#46; It may also be of value for planning and monitoring treatment&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">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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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Amat-Samaranch V&#44; Morales-M&#250;nera C&#44; Puig L&#44; Ro&#233; E&#46; Utilidad de la ecograf&#237;a cut&#225;nea para el diagn&#243;stico de la malformaci&#243;n venosa verrucosa&#46; Actas Dermosifiliogr&#46; 2021&#59;112&#58;958&#8211;961&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical images of verrucous venous malformations&#46; A&#44; Patient 1&#46; Verrucous erythematous violaceous plaque on the left ankle&#46; B&#44; Patient 2&#46; Erythematous&#44; violaceous&#44; hyperkeratotic plaques with satellite lesions and hemorrhagic superficial vesicles on the back of the right leg&#46; C&#44; Patient 3&#46; Erythematous&#44; violaceous plaques with a verrucous surface comprising hyperkeratotic areas and crusts involving the circumference of the distal region of the left leg&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Ultrasound images of verrucous venous malformations &#40;VVMs&#41;&#46; A&#44; Cutaneous ultrasound &#40;22&#160;MHz&#41; of the VVM in patient 1&#46; Thickened hyperechoic epidermis&#44; hypoechoic dermis&#44; hypoechoic vascular channels in the dermis and subcutaneous tissue&#44; and absence of color Doppler signal&#46; B&#44; Cutaneous ultrasound &#40;22&#160;MHz&#41; of VVM in patient 2&#46; Thickened&#44; hyperechoic epidermis with vascular channels in the superficial and deep dermis&#59; no color Doppler signal&#46; C&#44; Cutaneous ultrasound &#40;22&#160; MHz&#41; showing hyperechoic epidermis&#44; decreased dermal echogenicity&#44; and a loss of definition between the dermis and hypodermis&#46; Note the thickened&#44; heteroechoic subcutaneous tissue and absence of a color Doppler signal&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Clinical and ultrasound image of an angiokeratoma&#46; A&#44; Angiokeratoma circumscriptum&#46; Erythematous plaque with a hyperkeratotic surface&#44; clinically indistinguishable from verrucous venous malformation&#46; B&#44; Cutaneous ultrasound &#40;22&#160;MHz&#41;&#46; Epidermal hyperechogenicity with vascular channels limited to the papillary dermis&#59; no color Doppler signal&#46;</p>"
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