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Further investigation led to the diagnosis of essential thrombocythemia &#40;ET&#41; JAK2 negative&#44; confirmed by bone marrow examination&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Despite anticoagulation and optimized diuretic therapy&#44; renal function worsened and ascites persisted&#44; and the patient was proposed for liver transplantation&#46; Few weeks after transplantation&#44; spontaneous resolution of cutaneous lesions was noticed&#44; without relapse during 2 years of follow-up&#46; Concerning her hematologic disease&#44; she has been treated with hydroxyurea&#44; however with poor results&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Erythema annulare centrifugum &#40;EAC&#41; is a rare inflammatory skin disease presenting with erythematous papules or plaques that expand centrifugally with central clearing resulting in an annular shape&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1&#44;2</span></a> There are two types&#44; superficial and deep&#44; but this classification is not completely accepted and according to some authors the designation EAC should be reserved for the superficial type only&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">EAC has been considered a hypersensitivity reaction&#44; rather than a specific clinicopathologic entity&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">4</span></a> When a trigger is identified&#44; the treatment of the underlying condition mostly results in spontaneous resolution of the skin lesions&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">5</span></a> EAC has been associated with many different entities&#44; including infections&#44; malignant neoplasms&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">6</span></a> various autoimmune diseases<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">2&#44;3&#44;7</span></a> and drugs&#44;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">8</span></a> but its association with BCS or liver failure had not been previously described&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">BCS is a rare vascular liver disease resulting from the obstruction of the hepatic venous outflow tract that can result in liver failure&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">9</span></a> The most common underlying prothrombotic risk factor is a myeloproliferative disorder as ET&#44; although it is now recognized that almost half of patients have multiple underlying prothrombotic risk factors&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">10</span></a> Some chemical mediators released from activated platelets could explain some cutaneous findings associated with thrombocytosis such as erythromelalgia&#44; acute febril neutrophilic dermatosis and arterial thrombotic diseases<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">11</span></a> but not EAC&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In our case&#44; the skin lesion resolved without recurrence after liver transplantation&#46; On the other hand&#44; there was not an haematological improvement despite preconized therapy making the association between EAC and BCS&#47;liver disease more likely than with ET&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">To the best of our knowledge&#44; we report the first case of EAC probably associated with BCS in a patient with ET&#46; Our report shows a new association of EAC with a systemic disease&#44; thus emphasizing the importance of clinical and complementary evaluation in a patient with EAC&#44; as well as the importance of more studies to clarify the physiopathology of this clinical identity&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Borges AS&#44; Brasileiro A&#44; Santos S&#44; Saiote J&#46; Eritema anular centr&#237;fugo en un paciente con s&#237;ndrome de Budd-Chiari&#46; Actas Dermosifiliogr&#46; 2018&#59;109&#58;755&#8211;757&#46;</p>"
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Case and Research Letters
Erythema Annulare Centrifugum in a Patient With Budd-Chiari Syndrome
Eritema anular centrífugo en un paciente con síndrome de Budd-Chiari
A.S. Borgesa,
Autor para correspondencia
a.sofia.r.borges@gmail.com

Corresponding author.
, A. Brasileiroa, S. Santosb, J. Saioteb
a Unidad de Dermatología y Venereología, Hospital Santo António dos Capuchos, Centro Hospitalar de Lisboa Central, Lisboa, Portugal
b Unidad de Gastroenterología, Hospital Santo António dos Capuchos, Centro Hospitalar de Lisboa Central, Lisboa, Portugal
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based on the correlation between clinical and histological findings &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41; on hematoxylin and eosin stain&#46; Periodic acid-Schiff and Pearls colorations did not show remarkable findings&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Laboratory tests detected thrombocytosis&#59; negative viral hepatitis serology&#44; negative HIV&#44; CMV and EBV serology&#59; negative alfa fetoprotein&#59; negative autoantibodies &#40;ANA&#44; anti-sDNA&#44; antiphopholipid&#41;&#59; undetected rheumatoid factor&#59; normal levels of protein C&#44; S&#44; antithrombin II and homocysteine&#44; no resistance to activate protein C&#46; The search for prothrombin G20210A mutation was negative&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Doppler ultrasonography and computed tomography examination revealed thrombosis of the sus-hepatic veins and portal hypertension with the diagnostic of Budd Chiari-Syndrome &#40;BCS&#41;&#46; Further investigation led to the diagnosis of essential thrombocythemia &#40;ET&#41; JAK2 negative&#44; confirmed by bone marrow examination&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Despite anticoagulation and optimized diuretic therapy&#44; renal function worsened and ascites persisted&#44; and the patient was proposed for liver transplantation&#46; Few weeks after transplantation&#44; spontaneous resolution of cutaneous lesions was noticed&#44; without relapse during 2 years of follow-up&#46; Concerning her hematologic disease&#44; she has been treated with hydroxyurea&#44; however with poor results&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Erythema annulare centrifugum &#40;EAC&#41; is a rare inflammatory skin disease presenting with erythematous papules or plaques that expand centrifugally with central clearing resulting in an annular shape&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">1&#44;2</span></a> There are two types&#44; superficial and deep&#44; but this classification is not completely accepted and according to some authors the designation EAC should be reserved for the superficial type only&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">EAC has been considered a hypersensitivity reaction&#44; rather than a specific clinicopathologic entity&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">4</span></a> When a trigger is identified&#44; the treatment of the underlying condition mostly results in spontaneous resolution of the skin lesions&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">5</span></a> EAC has been associated with many different entities&#44; including infections&#44; malignant neoplasms&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">6</span></a> various autoimmune diseases<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">2&#44;3&#44;7</span></a> and drugs&#44;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">8</span></a> but its association with BCS or liver failure had not been previously described&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">BCS is a rare vascular liver disease resulting from the obstruction of the hepatic venous outflow tract that can result in liver failure&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">9</span></a> The most common underlying prothrombotic risk factor is a myeloproliferative disorder as ET&#44; although it is now recognized that almost half of patients have multiple underlying prothrombotic risk factors&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">10</span></a> Some chemical mediators released from activated platelets could explain some cutaneous findings associated with thrombocytosis such as erythromelalgia&#44; acute febril neutrophilic dermatosis and arterial thrombotic diseases<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">11</span></a> but not EAC&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In our case&#44; the skin lesion resolved without recurrence after liver transplantation&#46; On the other hand&#44; there was not an haematological improvement despite preconized therapy making the association between EAC and BCS&#47;liver disease more likely than with ET&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">To the best of our knowledge&#44; we report the first case of EAC probably associated with BCS in a patient with ET&#46; Our report shows a new association of EAC with a systemic disease&#44; thus emphasizing the importance of clinical and complementary evaluation in a patient with EAC&#44; as well as the importance of more studies to clarify the physiopathology of this clinical identity&#46;</p></span></span>"
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