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Letter to the Editor
Platypnea-orthodeoxia syndrome and Budd–Chiari syndrome: An unreported association
Síndrome de platipnea-ortodeoxia y síndrome de Budd-Chiari: una asociación inédita
Pablo Demelo-Rodríguez
Corresponding author
pbdemelo@hotmail.com

Corresponding author.
, Jorge del Toro-Cervera
Departamento de Medicina Interna, Hospital General Universitario Gregorio Marañón, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The platypnea-orthodeoxia syndrome &#40;POS&#41; is a very rare disease characterised by the presence of dyspnoea &#40;platypnea&#41; and arterial desaturation on standing position &#40;orthodeoxia&#41;&#44; which improves in the decubitus position&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">On the other hand&#44; the Budd&#8211;Chiari syndrome &#40;BCS&#41; is a condition characterised by the obstruction of the hepatic venous outflow in the absence of right heart failure or constrictive pericarditis&#46; Said obstruction may occur in the hepatic veins or suprahepatic inferior vena cava&#46; When the obstruction is caused by a thrombosis&#44; it is called primary BCS&#46; If it is caused by a tumour&#44; it is called secondary BCS&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">We presented the case of a 35-year-old female patient who&#44; 2 years before&#44; had been diagnosed with primary BCS during pregnancy &#40;week 11&#41;&#44; evidenced by ascites and dyspnoea&#46; The hepatic Doppler scanning showed thrombosis of the right and left suprahepatic veins and reduced flow in the middle suprahepatic vein&#46; Anticoagulation therapy was started with low-molecular-weight heparin&#44; and the patient gave birth on week 38 through vaginal delivery using forceps&#46; In the subsequent outpatient study&#44; the patient was diagnosed with polycythemia vera &#40;JAK2&#43;&#44; typical bone marrow biopsy and normal karyotype&#41;&#46; Therefore&#44; anticoagulation therapy was indefinitely maintained with acenocumarol&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">During the BCS progress control&#44; a year after the diagnosis&#44; there was still no flow in the right and left suprahepatic veins&#44; and the middle suprahepatic vein presented a large venous vessel compatible with collateral circulation&#46; The portal vein was normal and presented hepatopetal circulation &#40;at a speed of 23<span class="elsevierStyleHsp" style=""></span>cm&#47;s&#41;&#46; The liver and kidney functions were normal&#44; and the endoscopic test ruled out the presence of varicose veins&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient attended our practice because she had moderate-mild effort dyspnoea&#44; with predominance during orthostatism &#40;and&#44; in particular&#44; in some positions&#44; such as when leaning forward&#41; and improving in the decubitus position &#40;platypnea&#41;&#46; It had no other related symptoms&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The physical examination revealed hepatomegaly of 2 finger breadths&#44; without any other pathological findings&#46; The baseline saturation in oxygen was normal&#46; As to the complementary tests&#44; the electrocardiogram&#44; the cardiac stress test&#44; the spirometry and the diffusion test were normal&#46; A transthoracic echocardiogram revealed interauricular septal aneurysm with the presence of a permeable oval foramen &#40;POF&#41; with a right to left short circuit and high bubble load&#46; The rest of the results were normal&#46; Due to diagnostic suspicion&#44; the cardiac stress test was repeated and the patient was specifically asked to lean forward during the test&#46; In this way&#44; she showed a sudden decrease in the oxygen saturation at maximum effort &#40;up to a 78&#37;&#41; and a sudden decrease in the respiratory reserve from 70 &#40;6<span class="elsevierStyleHsp" style=""></span>min in the Bruce protocol&#41; to 48&#37; at maximum effort&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The oval foramen was closed by means of a percutaneous catheter procedure using an Amplatzer cribriform device of 25<span class="elsevierStyleHsp" style=""></span>mm&#44; with good results and no complications&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">First described in 1949&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> the POS results from a short circuit of non-oxygenated blood from the right auricle to the left auricle through an opening in the interauricular septum&#46; The POS is characterised by the presence of two components&#58; on the one hand&#44; an interauricular short circuit or <span class="elsevierStyleItalic">shunt</span> &#40;as an oval foramen or auricular communication&#41; or an intrapulmonary short circuit &#40;as the hepatopulmonary syndrome or pulmonary arteriovenous malformations&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> On the other hand&#44; the syndrome requires a functional component that favours the right-left short circuit when the patient moves from the decubitus position to orthostatism&#46; This could be a defect in the auricular septum or right auricle&#44; which increases blood flow from the inferior vena cava through the auricular defect&#46; In this regard&#44; various related diseases have been described&#44; such as pneumonectomy&#44; pulmonary hypertension or pleural effusion&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">4&#44;5</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Though the POF is present in up to one third of the general population&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> it almost never has clinical manifestations&#46; The most frequent presentation of the POS is a patient with an asymptomatic POF who&#44; after developing a disease which favours increased pressure in the right cavities&#44; has a clinical manifestation of dyspnoea and unsaturation with orthostatism&#46; In our patient&#44; the decrease in the flow of suprahepatic veins and the development of collaterals meet the requirements of a position-related redirection of the venous flow through the inferior vena cava to the auricular septum&#46; The presence of this disease and an oval foramen would be the two necessary requirements for the diagnosis of BCS-related POS&#46; There are currently no scientific publications on POS cases related to this condition&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">To date&#44; the subsequent progress of the patient has been favourable&#44; and dyspnoea related to position changes and mild efforts has not been observed&#46; Moderate-effort mild dyspnoea&#44; which is probably related to the baseline haematological disease&#44; has persisted&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Demelo-Rodr&#237;guez P&#44; del Toro-Cervera J&#46; S&#237;ndrome de platipnea-ortodeoxia y s&#237;ndrome de Budd-Chiari&#58; una asociaci&#243;n in&#233;dita&#46; Med Clin &#40;Barc&#41;&#46; 2015&#59;144&#58;94&#8211;95&#46;</p>"
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