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Although these antibodies do not impede prothrombin activity&#44; they lead to secondary hypoprothrombinaemia due to rapid clearance of the antigen&#8211;antibody complexes from the circulation&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The most common treatment of LAHS consists of steroid therapy together with other immunosuppressors &#40;cyclophosphamide&#44; azathioprine&#44; or rituximab&#41;&#46; The aim of treatment if to reduce the risk of bleeding and eliminate prothrombin inhibitor&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">We present the clinical case of a 37-year-old Bulgarian man&#44; with a history of lupus nephropathy diagnosed in his country in 2004&#46; Despite receiving treatment&#44; he had a poor clinical course&#44; requiring renal replacement therapy 7 years after diagnosis&#46; Two years later&#44; he attended our hospital to continue haemodialysis&#46; He was assessed by the rheumatology team and the haematology team for chronic thrombocytopenia&#44; for which he received immunosuppressive treatment with steroids&#44; immunoglobulins&#44; and rituximab&#44; as well as thrombopoietin-receptor agonists &#40;eltrombopag&#41;&#44; with a poor response&#46; He also had APS and was positive for anti-cardiolipin antibodies and anti-&#946;2-microglobulin antibodies&#46; This presented with several thrombotic events &#40;thrombosis of several vascular access devices&#41; and a cerebrovascular event in 2014&#44; for which he had been started on anticoagulant treatment&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was admitted to our department for an episode of general malaise and low-grade fever in the context of a possible central venous catheter bacteraemia&#46; He started broad-spectrum antibiotic treatment with a clear clinical improvement&#44; and it was decided to stop anticoagulation temporarily to allow a change of venous catheter&#46; During this period of withheld anticoagulation&#44; he experienced a transient ischaemic attack which presented as right-sided paraesthesia and reduced power&#44; with expressive aphasia&#46; It was therefore decided to re-start anticoagulation with heparin sodium&#46; Surprisingly&#44; brain magnetic resonance angiography showed&#44; along with old ischaemic lesions&#44; a significant subdural haematoma at the left cerebral convexity with mass effect in the parieto-occipital region &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Between the neurosurgery and neurology teams&#44; it was decided to stop anticoagulation and wait and see how he progressed clinically and radiologically&#46; Given the presence of thrombotic and haemorrhagic events&#44; a new coagulation study was requested&#46; This showed lupus anticoagulant&#44; elevated levels of anti-cardiolipin antibodies and anti-&#946;2-microglobulin antibodies&#44; and low coagulation factor II &#40;prothrombin&#41; activity&#44; which corrected with mixing&#44; confirming the presence of lupus anticoagulant-hypoprothrombinaemia syndrome &#40;<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Combined treatment was started with anticoagulation and immunosuppression with low dose steroids&#44; mycophenolic acid&#44; and rituximab 6-monthly&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The association of lupus anticoagulant with hypoprothrombinaemia is a rare syndrome&#44; infrequently described&#46; It is a syndrome that is more common in children and young adults&#44; and is commonly associated with viral infections and auto-immune diseases &#40;above all systemic lupus erythematosus&#41;&#44; although cases have also been described in association with drugs or tumours&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> LAHS is usually self-limiting when associated with viral infection&#44; whereas in those associated with auto-immune diseases&#44; relapse is common despite treatment&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">It is clinically characterised by haemorrhagic diathesis&#44; with epistaxes and ecchymoses being the most common types of bleed&#44; although cases have been described of haematuria&#44; gastrointestinal bleeding&#44; and intracranial haemorrhage&#44; amongst others<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Several cases have been described of LAHS with associated thrombosis&#44; in some cases multiple thromboses&#46; However&#44; those cases were mainly in the context of starting treatment against prothrombin inhibitor&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Treatment of LAHS is based on immunosuppression to avoid haemorrhagic events and to try to eliminate factor II-inhibitor&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> An increase in thrombotic events has been described in patients with LAHS&#44; as immunosuppression reduces inhibitor levels&#44; but not lupus anticoagulant levels&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> There are no guidelines indicating what the best treatment is for LAHS&#44; most treatment being based on corticoids with another immunosuppressor&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; in patients with lupus nephropathy with both thrombotic and haemorrhagic processes&#44; lupus anticoagulant-hypoprothrombinaemia syndrome must be suspected&#46; Diagnosis and treatment-based on immunosuppression to control the clinical manifestations of the disease are essential&#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 potential conflicts of interest related to the contents of this article&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Carre&#241;o-Tarragona G&#44; Morales E&#44; Jim&#233;nez-Herrero MC&#44; Cort&#233;s-Fornieles E&#44; Gutierrez E&#44; Praga M&#46; S&#237;ndrome de anticoagulante l&#250;pico-hipoprotrombinemia&#58; una extra&#241;a asociaci&#243;n en el lupus eritematoso sist&#233;mico&#46; Nefrologia&#46; 2016&#59;36&#58;186&#8211;188&#46;</p>"
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">70&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">83&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">96&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">94&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">98&nbsp;\t\t\t\t\t\t\n
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Letter to the Editor – Brief Case Reports
Lupus anticoagulant-hypoprothrombinemia syndrome: A rare association in systemic lupus erythematosus
Síndrome de anticoagulante lúpico-hipoprotrombinemia: una extraña asociación en el lupus eritematoso sistémico
Gonzalo Carreño-Tarragonaa, Enrique Moralesb,
Corresponding author
emoralesr@senefro.org

Corresponding author.
, María Carmen Jiménez-Herrerob, Elena Cortés-Fornielesb, Eduardo Gutierrezb, Manuel Pragab
a Servicio de Hematología, Hospital Universitario 12 de Octubre, Madrid, Spain
b Servicio de Nefrología, Hospital Universitario 12 de Octubre, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Lupus anticoagulant-hypoprothrombinaemia syndrome &#40;LAHS&#41; is a disorder characterised by the acquired deficit of coagulation factor II &#40;prothrombin&#41; together with the presence of lupus anticoagulant&#46; It is an extremely rare syndrome &#40;less than 100 cases described in the literature&#41;&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> in which there is a predisposition to bleeding&#44; unlike antiphospholipid syndrome &#40;APS&#41;&#44; which is characterised by an increased risk of thrombosis&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">The first case was described by Rapaport et al&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> in 1960&#44; but it was not until more than 20 years later that the study be Bajaj et al&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> demonstrated the presence of anti-prothrombin antibodies&#46; Although these antibodies do not impede prothrombin activity&#44; they lead to secondary hypoprothrombinaemia due to rapid clearance of the antigen&#8211;antibody complexes from the circulation&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The most common treatment of LAHS consists of steroid therapy together with other immunosuppressors &#40;cyclophosphamide&#44; azathioprine&#44; or rituximab&#41;&#46; The aim of treatment if to reduce the risk of bleeding and eliminate prothrombin inhibitor&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">We present the clinical case of a 37-year-old Bulgarian man&#44; with a history of lupus nephropathy diagnosed in his country in 2004&#46; Despite receiving treatment&#44; he had a poor clinical course&#44; requiring renal replacement therapy 7 years after diagnosis&#46; Two years later&#44; he attended our hospital to continue haemodialysis&#46; He was assessed by the rheumatology team and the haematology team for chronic thrombocytopenia&#44; for which he received immunosuppressive treatment with steroids&#44; immunoglobulins&#44; and rituximab&#44; as well as thrombopoietin-receptor agonists &#40;eltrombopag&#41;&#44; with a poor response&#46; He also had APS and was positive for anti-cardiolipin antibodies and anti-&#946;2-microglobulin antibodies&#46; This presented with several thrombotic events &#40;thrombosis of several vascular access devices&#41; and a cerebrovascular event in 2014&#44; for which he had been started on anticoagulant treatment&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was admitted to our department for an episode of general malaise and low-grade fever in the context of a possible central venous catheter bacteraemia&#46; He started broad-spectrum antibiotic treatment with a clear clinical improvement&#44; and it was decided to stop anticoagulation temporarily to allow a change of venous catheter&#46; During this period of withheld anticoagulation&#44; he experienced a transient ischaemic attack which presented as right-sided paraesthesia and reduced power&#44; with expressive aphasia&#46; It was therefore decided to re-start anticoagulation with heparin sodium&#46; Surprisingly&#44; brain magnetic resonance angiography showed&#44; along with old ischaemic lesions&#44; a significant subdural haematoma at the left cerebral convexity with mass effect in the parieto-occipital region &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Between the neurosurgery and neurology teams&#44; it was decided to stop anticoagulation and wait and see how he progressed clinically and radiologically&#46; Given the presence of thrombotic and haemorrhagic events&#44; a new coagulation study was requested&#46; This showed lupus anticoagulant&#44; elevated levels of anti-cardiolipin antibodies and anti-&#946;2-microglobulin antibodies&#44; and low coagulation factor II &#40;prothrombin&#41; activity&#44; which corrected with mixing&#44; confirming the presence of lupus anticoagulant-hypoprothrombinaemia syndrome &#40;<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Combined treatment was started with anticoagulation and immunosuppression with low dose steroids&#44; mycophenolic acid&#44; and rituximab 6-monthly&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The association of lupus anticoagulant with hypoprothrombinaemia is a rare syndrome&#44; infrequently described&#46; It is a syndrome that is more common in children and young adults&#44; and is commonly associated with viral infections and auto-immune diseases &#40;above all systemic lupus erythematosus&#41;&#44; although cases have also been described in association with drugs or tumours&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> LAHS is usually self-limiting when associated with viral infection&#44; whereas in those associated with auto-immune diseases&#44; relapse is common despite treatment&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">It is clinically characterised by haemorrhagic diathesis&#44; with epistaxes and ecchymoses being the most common types of bleed&#44; although cases have been described of haematuria&#44; gastrointestinal bleeding&#44; and intracranial haemorrhage&#44; amongst others<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Several cases have been described of LAHS with associated thrombosis&#44; in some cases multiple thromboses&#46; However&#44; those cases were mainly in the context of starting treatment against prothrombin inhibitor&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Treatment of LAHS is based on immunosuppression to avoid haemorrhagic events and to try to eliminate factor II-inhibitor&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> An increase in thrombotic events has been described in patients with LAHS&#44; as immunosuppression reduces inhibitor levels&#44; but not lupus anticoagulant levels&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> There are no guidelines indicating what the best treatment is for LAHS&#44; most treatment being based on corticoids with another immunosuppressor&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion&#44; in patients with lupus nephropathy with both thrombotic and haemorrhagic processes&#44; lupus anticoagulant-hypoprothrombinaemia syndrome must be suspected&#46; Diagnosis and treatment-based on immunosuppression to control the clinical manifestations of the disease are essential&#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 potential conflicts of interest related to the contents of this article&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Carre&#241;o-Tarragona G&#44; Morales E&#44; Jim&#233;nez-Herrero MC&#44; Cort&#233;s-Fornieles E&#44; Gutierrez E&#44; Praga M&#46; S&#237;ndrome de anticoagulante l&#250;pico-hipoprotrombinemia&#58; una extra&#241;a asociaci&#243;n en el lupus eritematoso sist&#233;mico&#46; Nefrologia&#46; 2016&#59;36&#58;186&#8211;188&#46;</p>"
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                  \t\t\t\t\tvoid\n
                  \t\t\t\t" class=""><thead title="thead"><tr title="table-row"><th class="td" title="table-head  " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Factor&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">Factor activity &#40;&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">Corrected factor activity &#40;SynthAFax<span class="elsevierStyleSup">&#174;</span>&#41; &#40;&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th><th class="td" title="table-head  " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">Mixed factor activity &#40;&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</th></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor II&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">25&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">70&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor V&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">102&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor VII&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">136&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor X&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">114&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor VIII&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">16&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">83&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor IX&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">6&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">96&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor XI&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">8&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">94&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry  " align="left" valign="top">Factor XII&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">7&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">98&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="table-entry  " align="char" valign="top">&#8211;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></tbody></table>
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ISSN: 20132514
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Idiomas
Nefrología (English Edition)