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<article xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" article-type="letter" dtd-version="1.1" specific-use="sps-1.9" xml:lang="pt">
  <front>
    <journal-meta>
      <journal-id journal-id-type="nlm-ta">Arq Bras Cardiol</journal-id>
      <journal-id journal-id-type="publisher-id">abc</journal-id>
      <journal-title-group>
        <journal-title>Arquivos Brasileiros de Cardiologia</journal-title>
        <abbrev-journal-title abbrev-type="publisher">Arq. Bras. Cardiol.</abbrev-journal-title>
      </journal-title-group>
      <issn pub-type="ppub">0066-782X</issn>
      <issn pub-type="epub">1678-4170</issn>
      <publisher>
        <publisher-name>Sociedade Brasileira de Cardiologia - SBC</publisher-name>
      </publisher>
    </journal-meta>
    <article-meta>
      <article-id specific-use="scielo-v3" pub-id-type="publisher-id">QF83GYnytbNDvVkrQYJms9L</article-id>
      <article-id specific-use="scielo-v2" pub-id-type="publisher-id">S0066-782X2022000300659</article-id>
      <article-id pub-id-type="publisher-id">abc.20210409</article-id>
      <article-id pub-id-type="doi">10.36660/abc.20210409</article-id>
      <article-categories>
        <subj-group subj-group-type="heading">
          <subject>Carta Cient&#237;fica</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title>Uma Causa Rara de Hipoxemia ap&#243;s Cirurgia Ortop&#233;dica num Doente Idoso</article-title>
      </title-group>
      <contrib-group>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0002-6954-4925</contrib-id>
          <name>
            <surname>Carvalho</surname>
            <given-names>Pedro</given-names>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Meireles</surname>
            <given-names>Daniela</given-names>
          </name>
          <xref ref-type="aff" rid="aff2">
            <sup>2</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0001-7198-771X</contrib-id>
          <name>
            <surname>Martins</surname>
            <given-names>Jos&#233; Lu&#237;s</given-names>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Costa</surname>
            <given-names>Marco</given-names>
          </name>
          <xref ref-type="aff" rid="aff3">
            <sup>3</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Neves</surname>
            <given-names>Ana Briosa</given-names>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
      </contrib-group>
      <aff id="aff1">
        <label>1</label>
        <institution content-type="orgname">Centro Hospitalar do Baixo Vouga</institution>
        <institution content-type="orgdiv1">Cardiology Department</institution>
        <addr-line>
          <named-content content-type="city">Aveiro</named-content>
        </addr-line>
        <country country="PT">Portugual</country>
        <institution content-type="original">Centro Hospitalar do Baixo Vouga &#8211; Cardiology Department, Aveiro &#8211; Portugual</institution>
      </aff>
      <aff id="aff2">
        <label>2</label>
        <institution content-type="orgname">Centro Hospitalar do Baixo Vouga</institution>
        <institution content-type="orgdiv1">Internal Medicine Department</institution>
        <addr-line>
          <named-content content-type="city">Aveiro</named-content>
        </addr-line>
        <country country="PT">Portugal</country>
        <institution content-type="original">Centro Hospitalar do Baixo Vouga &#8211; Internal Medicine Department,Aveiro &#8211; Portugal</institution>
      </aff>
      <aff id="aff3">
        <label>3</label>
        <institution content-type="orgname">Centro Hospitalar Universit&#225;rio de Coimbra</institution>
        <institution content-type="orgdiv1">Cardiology Department</institution>
        <addr-line>
          <named-content content-type="city">Coimbra</named-content>
        </addr-line>
        <country country="PT">Portugal</country>
        <institution content-type="original">Centro Hospitalar Universit&#225;rio de Coimbra &#8211; Cardiology Department, Coimbra &#8211; Portugal</institution>
      </aff>
      <author-notes>
        <corresp id="c01"> Correspond&#234;ncia: Pedro Carvalho &#8226; Centro Hospitalar do Baixo Vouga EPE &#8211; Cardiologia <italic>&#8211;</italic> Avenida Doutor Artur Ravara, Aveiro &#8211; Portugal. C&#243;digo postal: 3810-501 E-mail: <email>carvalho.pmt@gmail.com</email> </corresp>
        <fn fn-type="con">
          <p>Contribui&#231;&#227;o dos autores</p>
          <p>Concep&#231;&#227;o e desenho da pesquisa e Revis&#227;o cr&#237;tica do manuscrito quanto ao conte&#250;do intelectual importante: Carvalho P, Meireles D, Martins J, Costa MA, Briosa A; Obten&#231;&#227;o de dados e An&#225;lise e interpreta&#231;&#227;o dos dados: Carvalho P, Meireles D, Martins J, Costa MA; Reda&#231;&#227;o do manuscrito: Carvalho P.</p>
        </fn>
        <fn fn-type="conflict">
          <p>Potencial conflito de interesse</p>
          <p>N&#227;o h&#225; conflito com o presente artigo</p>
        </fn>
      </author-notes>
      <pub-date date-type="pub" publication-format="electronic">
        <day>18</day>
        <month>03</month>
        <year>2022</year>
      </pub-date>
      <pub-date date-type="collection" publication-format="electronic">
        <month>03</month>
        <year>2022</year>
      </pub-date>
      <volume>118</volume>
      <issue>3</issue>
      <fpage>659</fpage>
      <lpage>662</lpage>
      <history>
        <date date-type="received">
          <day>12</day>
          <month>05</month>
          <year>2021</year>
        </date>
        <date date-type="rev-recd">
          <day>05</day>
          <month>08</month>
          <year>2021</year>
        </date>
        <date date-type="accepted">
          <day>08</day>
          <month>09</month>
          <year>2021</year>
        </date>
      </history>
      <permissions>
        <license license-type="open-access" xlink:href="https://creativecommons.org/licenses/by/4.0/" xml:lang="en">
          <license-p> This is an Open Access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. </license-p>
        </license>
      </permissions>
      <kwd-group xml:lang="pt">
        <kwd>Forame Oval Patente</kwd>
        <kwd>Hip&#243;xia</kwd>
        <kwd>Dispositivos de Oclus&#227;o Vascular</kwd>
      </kwd-group>
      <counts>
        <fig-count count="6"/>
        <table-count count="2"/>
        <equation-count count="0"/>
        <ref-count count="8"/>
        <page-count count="4"/>
      </counts>
    </article-meta>
  </front>
  <body>
    <sec sec-type="intro">
      <title>Introdu&#231;&#227;o</title>
      <p>V&#225;rias doen&#231;as podem causar hipoxemia p&#243;s-operat&#243;ria, especialmente em pacientes idosos. Entretanto, um novo shunt card&#237;aco &#233; uma complica&#231;&#227;o muito rara e inesperada nesse cen&#225;rio. Este estudo relata um caso de hipoxemia refrat&#225;ria ap&#243;s cirurgia ortop&#233;dica devido a um shunt da direita para a esquerda via um forame oval patente (FOP).</p>
    </sec>
    <sec sec-type="cases">
      <title>Relato de caso</title>
      <p>Um paciente do sexo masculino de 71 anos foi submetido a cirurgia eletiva para artroplastia do quadril esquerdo, com anestesia locorregional. Seu hist&#243;rico m&#233;dico inclu&#237;a obesidade, hipertens&#227;o, diabetes mellitus e um acidente vascular cerebral. Ele n&#227;o tinha hist&#243;rico de doen&#231;a cardiopulmonar.</p>
      <p>O primeiro dia do p&#243;s-operat&#243;rio foi complicado por obstru&#231;&#227;o intestinal ( <xref ref-type="fig" rid="f01">Figura 1</xref> ). A alimenta&#231;&#227;o foi reiniciada quatro dias depois, mas a distens&#227;o abdominal e a redu&#231;&#227;o dos movimentos perist&#225;lticos persistiram. No 15&#186; dia de p&#243;s-operat&#243;rio, o paciente apresentou hipoxemia refrat&#225;ria grave, com uma satura&#231;&#227;o de O<sub>2</sub> (O<sub>2</sub>sat) de 75%, chegando apenas a 86% com oxigenoterapia de alto fluxo (F<sub>i</sub>O<sub>2</sub> 90-100%). Apesar disso, o paciente estava calmo, sem apresentar sinais de desconforto respirat&#243;rio. A press&#227;o arterial era 110/75 mmHg, a frequ&#234;ncia card&#237;aca era 76 bpm e a temperatura era 36 &#186;C. A ausculta&#231;&#227;o card&#237;aca e a pulmonar estavam normais. N&#227;o havia distens&#227;o venosa jugular, edema perif&#233;rico ou cianose.</p>
      <p>
        <fig id="f01">
          <label>Figura 1</label>
          <caption>
            <title>
              <italic>Radiografia abdominal demonstrando distens&#227;o abdominal.</italic>
            </title>
          </caption>
          <graphic xlink:href="1678-4170-abc-118-03-0659-10409-gf01.tif"/></fig>
      </p>
      <p>A gasometria arterial confirmou hipoxemia grave, com uma pO<sub>2</sub> de 38 mmHg em F<sub>i</sub>O<sub>2</sub> de 28%, chegando apenas a 49 mmHg em F<sub>i</sub>O<sub>2</sub> 100%. O hemograma estava normal, exceto pelo alto n&#237;vel de d&#237;meros-D (1608 ng/mL). O eletrocardiograma e o ecocardiograma &#224; beira do leito estavam normais. O paciente foi submetido a uma angiotomografia computadorizada arterial de t&#243;rax que n&#227;o revelou sinais de embolia pulmonar ou doen&#231;a pulmonar parenquimatosa significativa. Nos dias seguintes, a O<sub>2</sub>sat foi mantida em 85-89% apesar da c&#226;nula nasal de alto fluxo de oxig&#234;nio, independentemente de o paciente estar em p&#233;, em posi&#231;&#227;o supina ou em dec&#250;bito lateral esquerdo.</p>
      <p>Foi realizada uma cintilografia de ventila&#231;&#227;o e perfus&#227;o pulmonar (V/Q), demonstrando a aus&#234;ncia de desequil&#237;brio V/Q, mas revelando capta&#231;&#227;o do marcador pelo c&#233;rebro e pelo rim, sugerindo shunt D-E ( <xref ref-type="fig" rid="f02">Figura 2A</xref> ). O ecocardiograma transesof&#225;gico (ETE) revelou um aneurisma do septo interatrial e um FOP com um grande shunt D-E em repouso, vis&#237;vel por Doppler colorido e inje&#231;&#227;o de solu&#231;&#227;o salina agitada ( <xref ref-type="fig" rid="f02">Figura 2B</xref> ). Nas an&#225;lises de imagens de tomografia computadorizada (TC), verificou-se que a distens&#227;o intestinal havia causado a eleva&#231;&#227;o do hemidiafragma esquerdo, alterando o eixo da veia cava inferior supra-hep&#225;tica e a posi&#231;&#227;o do cora&#231;&#227;o (e, consequentemente, a posi&#231;&#227;o do septo interatrial) horizontalmente ( <xref ref-type="fig" rid="f02">Figura 2C</xref> ). Em imagem de contraste, observou-se a opacifica&#231;&#227;o inicial das c&#226;maras card&#237;acas esquerdas.</p>
      <p>
        <fig id="f02">
          <label>Figura 2</label>
          <caption>
            <title>
              <italic>Painel A: Cintilografia de V/Q demonstrando capta&#231;&#227;o, pelo c&#233;rebro e pelo rim, de macroagregado de albumina 99m Tc; Painel B: ETE demonstrando um aneurisma do septo interatrial e FOP, com um grande shunt D-E em repouso, vis&#237;vel por Doppler colorido e inje&#231;&#227;o de solu&#231;&#227;o salina agitada; Painel C: Imagens de TC demonstrando a eleva&#231;&#227;o do hemidiafragma esquerdo* alterando o eixo da veia cava inferior supra-hep&#225;tica* e a posi&#231;&#227;o do cora&#231;&#227;o* horizontalmente.</italic>
            </title>
          </caption>
          <graphic xlink:href="1678-4170-abc-118-03-0659-10409-gf02.tif"/></fig>
      </p>
      <p>No 32&#186; dia do p&#243;s-operat&#243;rio, o paciente foi submetido a cateterismo do cora&#231;&#227;o direito. A press&#227;o arterial pulmonar (PAP) estava normal (sist&#243;lica: 34 mmHg; diast&#243;lica: 9 mmHg; m&#233;dia: 20 mmHg). Foi realizado um teste de oclus&#227;o inflando-se um bal&#227;o dilatador pelo FOP ( <xref ref-type="fig" rid="f03">Figura 3A</xref> ) &#8211; a O<sub>2</sub>sat sist&#234;mica aumentou de 77% para 95% e a pO<sub>2</sub> arterial aumentou de 41 para 70 mmHg com ar ambiente, mantendo um PAP normal ( <xref ref-type="table" rid="t1">Tabela 1</xref> ). A oclus&#227;o foi realizada com um dispositivo oclusivo Amplatzer&#174; ASD de 14 mm ( <xref ref-type="fig" rid="f03">Figura 3B</xref> ). No ETE de acompanhamento, n&#227;o foi observado vazamento residual. Posteriormente o paciente recebeu alta em terapia antiplaquet&#225;ria dupla, com uma O<sub>2</sub>sat de 98% em ar ambiente. O tratamento com clopidogrel foi interrompido um m&#234;s ap&#243;s o procedimento. Ele permaneceu assintom&#225;tico no acompanhamento de um ano.</p>
      <p>
        <fig id="f03">
          <label>Figura 3</label>
          <caption>
            <title>
              <italic>Painel A: Teste de oclus&#227;o do FOP realizado por bal&#227;o dilatador no FOP; Painel B: Coloca&#231;&#227;o de um dispositivo oclusivo Amplatzer&#174; ASD de 14 mm.</italic>
            </title>
          </caption>
          <graphic xlink:href="1678-4170-abc-118-03-0659-10409-gf03.tif"/></fig>
      </p>
      <p>
        <table-wrap id="t1">
          <label>Tabela 1</label>
          <caption>
            <title>Gasometria realizada durante cateterismo do cora&#231;&#227;o direito</title>
          </caption>
          <table frame="hsides" rules="groups">
            <colgroup>
              <col/>
              <col/>
              <col/>
              <col/>
              <col/>
            </colgroup>
            <thead>
              <tr>
                <th align="left" rowspan="3" style="font-weight:normal"> </th>
                <th colspan="2">Antes da oclus&#227;o do bal&#227;o</th>
                <th colspan="2">Ap&#243;s a oclus&#227;o do bal&#227;o</th>
              </tr>
              <tr>
                <th colspan="4" rowspan="1">
                  <hr/>
                </th>
              </tr>
              <tr>
                <th>Art&#233;ria pulmonar</th>
                <th>Art&#233;ria radial</th>
                <th>Art&#233;ria pulmonar</th>
                <th>Art&#233;ria radial</th>
              </tr>
            </thead>
            <tbody>
              <tr>
                <td>pH</td>
                <td align="center">7,46</td>
                <td align="center">7,47</td>
                <td align="center">7,41</td>
                <td align="center">7,41</td>
              </tr>
              <tr>
                <td>
                  <bold>pCO</bold>
                  <sub>
                    <bold>2</bold>
                  </sub>
                  <bold>(mmHg)</bold>
                </td>
                <td align="center">30</td>
                <td align="center">26</td>
                <td align="center">32</td>
                <td align="center">29</td>
              </tr>
              <tr>
                <td>
                  <bold>pO</bold>
                  <sub>
                    <bold>2</bold>
                  </sub>
                  <bold>(mmHg)</bold>
                </td>
                <td align="center">25</td>
                <td align="center">
                  <bold>41</bold>
                </td>
                <td align="center">32</td>
                <td align="center">
                  <bold>70</bold>
                </td>
              </tr>
              <tr>
                <td>
                  <bold>O</bold>
                  <sub>
                    <bold>2</bold>
                  </sub>
                  <bold>sat (%)</bold>
                </td>
                <td align="center">52</td>
                <td align="center">
                  <bold>83</bold>
                </td>
                <td align="center">65</td>
                <td align="center">
                  <bold>95</bold>
                </td>
              </tr>
              <tr>
                <td>
                  <bold>HCO</bold>
                  <sub>
                    <bold>3-</bold>
                  </sub>
                  <bold>(mmol/L)</bold>
                </td>
                <td align="center">21</td>
                <td align="center">19</td>
                <td align="center">20</td>
                <td align="center">20</td>
              </tr>
            </tbody>
          </table>
        </table-wrap>
      </p>
    </sec>
    <sec sec-type="discussion">
      <title>Discuss&#227;o</title>
      <p>Estima-se que a preval&#234;ncia de FOP na popula&#231;&#227;o em geral seja de ~25%.<sup><xref ref-type="bibr" rid="B1">1</xref></sup> Na maioria dos casos, o shunt interatrial &#233; hemodinamicamente insignificante. Entretanto, em circunst&#226;ncias raras, um shunt D-E atrav&#233;s de um FOP pode causar desoxigena&#231;&#227;o arterial clinicamente significativa misturando sangue venoso e arterial. Esses pacientes geralmente apresentam s&#237;ndrome de platipneia-ortodeoxia, uma doen&#231;a rara caracterizada por dispneia e desoxigena&#231;&#227;o arterial induzida pela posi&#231;&#227;o em p&#233; e geralmente aliviada quando o paciente assume a posi&#231;&#227;o supino.<sup><xref ref-type="bibr" rid="B2">2</xref></sup></p>
      <p>A ocorr&#234;ncia de shunt interatrial D-E geralmente est&#225; associada &#224; hipertens&#227;o pulmonar espont&#226;nea ou induzida. A ocorr&#234;ncia desse shunt com press&#227;o arterial pulmonar normal &#233; muito rara, mas j&#225; foi descrita em relatos de casos anteriores. Isso ocorre com o fluxo sangu&#237;neo preferencial passando da veia cava inferior para o &#225;trio esquerdo, atrav&#233;s do FOP, mesmo na aus&#234;ncia de um gradiente de press&#227;o interartrial. Uma v&#225;lvula de Eust&#225;quio proeminente e uma altera&#231;&#227;o na anatomia da c&#226;mara direita podem ser fatores que contribuem para isso. Essa s&#237;ndrome foi descrita em pacientes com problemas mec&#226;nicos que causam deformidade atrial ou do septo, como a cifoescoliose,<sup><xref ref-type="bibr" rid="B3">3</xref></sup> doen&#231;a pulmonar restritiva, pneumonectomia pr&#233;via,<sup><xref ref-type="bibr" rid="B4">4</xref></sup> efus&#227;o pleural,<sup><xref ref-type="bibr" rid="B5">5</xref></sup> paralisia e ascens&#227;o diafragm&#225;tica,<sup><xref ref-type="bibr" rid="B6">6</xref></sup> aneurisma da aorta ascendente<sup><xref ref-type="bibr" rid="B3">3</xref></sup> ou p&#243;s-toracotomia.<sup><xref ref-type="bibr" rid="B7">7</xref></sup> Nesses casos, a rela&#231;&#227;o anat&#244;mica entre o septo atrial e a veia cava inferior foi alterada, facilitando o redirecionamento do fluxo de sangue dessaturado atrav&#233;s do FOP.</p>
      <p>O hist&#243;rico de sintomas pode ser curto e pode haver um aparecimento agudo, com piora r&#225;pida em alguns dias. Propriamente dito, o diagn&#243;stico de uma s&#237;ndrome de desoxigena&#231;&#227;o arterial geralmente &#233; um diagn&#243;stico &#8220;por exclus&#227;o&#8221;.<sup><xref ref-type="bibr" rid="B2">2</xref></sup> Causas comuns de hipoxemia aguda, tais como pneumonia, insufici&#234;ncia card&#237;aca aguda, embolia pulmonar ou outra doen&#231;a pulmonar estrutural, devem ser exclu&#237;das primeiramente. No caso desse estudo, o ecocardiograma transtor&#225;cico n&#227;o havia conseguido realizar o diagn&#243;stico. A primeira dica veio da cintilografia pulmonar de V/Q solicitada para excluir a embolia pulmonar ou outros desequil&#237;brios de V/Q, que revelou capta&#231;&#227;o do marcador pelo c&#233;rebro e pelo rim, um achado que &#233; diagn&#243;stico de shunt D-E.<sup><xref ref-type="bibr" rid="B8">8</xref></sup> O ETE confirmou o shunt D-E pela passagem do fluxo sangu&#237;neo da veia cava inferior para o FOP. A an&#225;lise das imagens de TC revelou que a distens&#227;o abdominal devido &#224; obstru&#231;&#227;o intestinal p&#243;s-operat&#243;ria tinha causado a eleva&#231;&#227;o do diafragma e a deforma&#231;&#227;o card&#237;aca, que, neste caso, foram respons&#225;veis pelo fluxo sangu&#237;neo. Depois de uma extensa revis&#227;o da literatura, identificou-se que este &#233; o primeiro caso relatado de s&#237;ndrome de desoxigena&#231;&#227;o arterial devido ao FOP nessas circunst&#226;ncias. Outra caracter&#237;stica exclusiva deste caso foi a grave hipoxemia em posi&#231;&#227;o supina, ao contr&#225;rio do al&#237;vio t&#237;pico da desoxigena&#231;&#227;o em posi&#231;&#227;o supina de pacientes com s&#237;ndrome de platipneia-ortodeoxia. Isso sugere que a deforma&#231;&#227;o anat&#244;mica que leva ao fluxo sangu&#237;neo n&#227;o dependia da posi&#231;&#227;o corporal.</p>
      <p>Uma poss&#237;vel limita&#231;&#227;o da documenta&#231;&#227;o deste caso foi a n&#227;o realiza&#231;&#227;o de uma gasometria completa em posi&#231;&#245;es corporais diferentes. Isso aconteceu pelo fato de a hipoxemia grave j&#225; ter sido documentada em dec&#250;bito sem mudan&#231;a significativa na oximetria de pulso em posi&#231;&#227;o sentada ou em p&#233;, e, portanto pareceu ser clinicamente desnecess&#225;rio realizar mais pun&#231;&#245;es da art&#233;ria radial no momento.</p>
    </sec>
    <sec sec-type="conclusions">
      <title>Conclus&#227;o</title>
      <p>O presente caso ilustra o diagn&#243;stico e o tratamento bem-sucedido de uma causa rara de hipoxemia e destaca os mecanismos causadores de fluxo card&#237;aco anormal e oxigena&#231;&#227;o deficiente com shunts D-E, que, em casos raros, podem ocorrer apesar das press&#245;es normais nas c&#226;maras.</p>
    </sec>
  </body>
  <back>
    <ref-list>
      <title>Refer&#234;ncias</title>
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        <label>1</label>
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              <surname>Pristipino</surname>
              <given-names>C</given-names>
            </name>
            <name>
              <surname>Sievert</surname>
              <given-names>H</given-names>
            </name>
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    <fn-group>
      <fn fn-type="other">
        <p>Vincula&#231;&#227;o acad&#234;mica</p>
        <p>N&#227;o h&#225; vincula&#231;&#227;o deste estudo a programas de p&#243;s-gradua&#231;&#227;o.</p>
      </fn>
    </fn-group>
    <fn-group>
      <fn fn-type="supported-by">
        <p>Fontes de financiamento: O presente estudo n&#227;o teve fontes de financiamento externas.</p>
      </fn>
    </fn-group>
  </back>
  <sub-article article-type="translation" id="TRen" xml:lang="en">
    <front-stub>
      <article-categories>
        <subj-group subj-group-type="heading">
          <subject>Research Letter</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title>An Unusual Cause of Hypoxemia after Orthopedic Surgery on an Elderly Patient</article-title>
      </title-group>
      <contrib-group>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0002-6954-4925</contrib-id>
          <name>
            <surname>Carvalho</surname>
            <given-names>Pedro</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Meireles</surname>
            <given-names>Daniela</given-names>
          </name>
          <xref ref-type="aff" rid="aff2001">
            <sup>2</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0001-7198-771X</contrib-id>
          <name>
            <surname>Martins</surname>
            <given-names>Jos&#233; Lu&#237;s</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Costa</surname>
            <given-names>Marco</given-names>
          </name>
          <xref ref-type="aff" rid="aff3001">
            <sup>3</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Neves</surname>
            <given-names>Ana Briosa</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
      </contrib-group>
      <aff id="aff1001">
        <label>1</label>
        <country country="PT">Portugual</country>
        <institution content-type="original">Centro Hospitalar do Baixo Vouga &#8211; Cardiology Department, Aveiro &#8211; Portugual</institution>
      </aff>
      <aff id="aff2001">
        <label>2</label>
        <country country="PT">Portugal</country>
        <institution content-type="original">Centro Hospitalar do Baixo Vouga &#8211; Internal Medicine Department, Aveiro &#8211; Portugal</institution>
      </aff>
      <aff id="aff3001">
        <label>3</label>
        <country country="PT">Portugal</country>
        <institution content-type="original">Centro Hospitalar Universit&#225;rio de Coimbra &#8211; Cardiology Department, Coimbra &#8211; Portugal</institution>
      </aff>
      <author-notes>
        <corresp id="c01001">Mailing Address: Pedro Carvalho &#8226; Centro Hospitalar do Baixo Vouga EPE &#8211; Cardiologia <italic>&#8211;</italic> Avenida Doutor Artur Ravara, Aveiro &#8211; Portugal. Mailing adress: 3810-501 E-mail: carvalho.pmt@gmail.com</corresp>
        <fn fn-type="con">
          <p>Author Contributions</p>
          <p>Conception and design of the research and Critical revision of the manuscript for intellectual content: Carvalho P, Meireles D, Martins J, Costa MA, Briosa A; Acquisition of data and Analysis and interpretation of the data: Carvalho P, Meireles D, Martins J, Costa MA; Writing of the manuscript: Carvalho P.</p>
        </fn>
        <fn fn-type="conflict">
          <p>Potential Conflict of Interest</p>
          <p>No potential conflict of interest relevant to this article was reported.</p>
        </fn>
      </author-notes>
      <kwd-group xml:lang="en">
        <kwd>Foramen Ovale, Patent</kwd>
        <kwd>Hypoxia</kwd>
        <kwd>Vascular Closure Devices</kwd>
      </kwd-group>
    </front-stub>
    <body>
      <sec sec-type="intro">
        <title>Introduction</title>
        <p>Various conditions can cause post-operative hypoxemia, especially in elderly patients. However, a new symptomatic cardiac shunt is a very rare and unexpected complication in this setting. This study presents a case of refractory hypoxemia after orthopedic surgery due to right-to-left (R-L) shunt via a patent foramen ovale (PFO).</p>
      </sec>
      <sec sec-type="cases">
        <title>Case Report</title>
        <p>A 71-year-old male underwent elective left hip replacement surgery, under loco-regional anesthesia. His medical history included obesity, hypertension, diabetes mellitus, and a stroke. He had no history of cardiopulmonary disease.</p>
        <p>The first postoperative day was complicated by ileus ( <xref ref-type="fig" rid="f01001">Figure 1</xref> ). Diet was restarted four days later, but abdominal distention and reduced bowel movements persisted. On postoperative day 15, the patient presented severe refractory hypoxemia, with an O<sub>2</sub>saturation (O<sub>2</sub>sat) of 75%, improving only to 86% on high-flow oxygen therapy (F<sub>i</sub>O<sub>2</sub>90-100%). Despite this, he was calm, showing no signs of respiratory distress. Blood pressure was 110/75 mmHg, heart rate was 76 bpm, and temperature was 36&#186;C. Cardiac and pulmonary auscultation were normal. There was no jugular venous distension, peripheral edema, or cyanosis.</p>
        <p>
          <fig id="f01001">
            <label>Figure 1</label>
            <caption>
              <title>
                <italic>Abdominal radiography demonstrating bowel distention.</italic>
              </title>
            </caption>
            <graphic xlink:href="1678-4170-abc-118-03-0659-10409-gf01-en.tif"/></fig>
        </p>
        <p>Arterial blood gas analysis confirmed severe hypoxemia, with a pO<sub>2</sub>of 38 mmHg on F<sub>i</sub>O<sub>2</sub>of 28%, improving only to 49 mmHg on F<sub>i</sub>O<sub>2</sub>100%. Blood analysis was unremarkable, except for elevated d-dimers (1608 ng/mL). Electrocardiogram and bedside echocardiogram were normal. The patient underwent chest computed tomography (CT) angiography, which showed no signs of pulmonary embolism or significant parenchymal lung disease. The following days, he maintained an O<sub>2</sub>sat of 85-89%, despite high-flow oxygen nasal cannula, regardless of upright, supine, or left lateral decubitus body position.</p>
        <p>A ventilation/perfusion (VQ) lung scan was performed, demonstrating the absence of VQ imbalance, but revealing a brain and kidney uptake of tracer, suggesting an R-L shunt ( <xref ref-type="fig" rid="f02001">Figure 2A</xref> ). Transoesophageal echocardiography (TOE) revealed an interatrial septal aneurysm and a PFO with a large resting R-L shunt visible by color Doppler and agitated saline injection ( <xref ref-type="fig" rid="f02001">Figure 2B</xref> ). Upon review of CT images, bowel distention was verified to have caused left hemidiaphragm elevation, changing the supra-hepatic inferior vena cava axis and heart position (and, consequently, the interatrial septal position) horizontally. ( <xref ref-type="fig" rid="f02001">Figure 2C</xref> ) On contrast imaging, early opacification of the left cardiac chambers was noted.</p>
        <p>
          <fig id="f02001">
            <label>Figure 2</label>
            <caption>
              <title>
                <italic>Panel A: V/Q scan demonstrating brain and kidney uptake of 99mTc-macroaggregated albumin; Panel B: TOE demonstrating PFO and interatrial septal aneurysm, with a large resting R-L shunt visible by color Doppler and agitated saline injection; Panel C: CT imaging demonstrating left hemidiaphragm elevation*, changing the supra-hepatic inferior vena cava axis* and the heart position* horizontally.</italic>
              </title>
            </caption>
            <graphic xlink:href="1678-4170-abc-118-03-0659-10409-gf02-en.tif"/></fig>
        </p>
        <p>On postoperative day 32 the patient underwent right heart catheterization. Pulmonary artery pressures (PAP) were normal (systolic: 34mmHg; diastolic: 9mmHg; mean: 20mmHg). An occlusion test was performed by inflating a sizing balloon on the PFO ( <xref ref-type="fig" rid="f03001">Figure 3A</xref> ) - systemic O<sub>2</sub>sat increased from 77% to 95%, and arterial pO<sub>2</sub>increased from 41 to 70 mmHg in room air, while maintaining a normal PAP ( <xref ref-type="table" rid="t1001">Table 1</xref> ). Closure was performed with a 14 mm Amplatzer&#174; ASD occluder device ( <xref ref-type="fig" rid="f03001">Figure 3B</xref> ). On follow-up TOE, no residual leak was noted. The patient was later discharged on dual antiplatelet therapy, with an O<sub>2</sub>sat of 98% in room air. Clopidogrel was stopped 1 month after the procedure. He remains asymptomatic at 1-year follow-up.</p>
        <p>
          <fig id="f03001">
            <label>Figure 3</label>
            <caption>
              <title>
                <italic>Panel A: PFO occlusion test performed by inflating sizing balloon on the PFO; Panel B: Deployment of a 14 mm Amplatzer&#174; ASD occluder device.</italic>
              </title>
            </caption>
            <graphic xlink:href="1678-4170-abc-118-03-0659-10409-gf03-en.tif"/></fig>
        </p>
        <p>
          <table-wrap id="t1001">
            <label>Table 1</label>
            <caption>
              <title>Blood gas analysis during right heart catheterization</title>
            </caption>
            <table frame="hsides" rules="groups">
              <colgroup>
                <col/>
                <col/>
                <col/>
                <col/>
                <col/>
              </colgroup>
              <thead>
                <tr>
                  <th align="left" rowspan="3" style="font-weight:normal"> </th>
                  <th colspan="2">Before balloon occlusion</th>
                  <th colspan="2">After balloon occlusion</th>
                </tr>
                <tr>
                  <th colspan="4" rowspan="1">
                    <hr/>
                  </th>
                </tr>
                <tr>
                  <th>Pulmonary artery</th>
                  <th>Radial artery</th>
                  <th>Pulmonary artery</th>
                  <th>Radial artery</th>
                </tr>
              </thead>
              <tbody>
                <tr>
                  <td>pH</td>
                  <td align="center">7.46</td>
                  <td align="center">7.47</td>
                  <td align="center">7.41</td>
                  <td align="center">7.41</td>
                </tr>
                <tr>
                  <td>
                    <bold>pCO</bold>
                    <sup>
                      <bold>2</bold>
                    </sup>
                    <bold>(mmHg)</bold>
                  </td>
                  <td align="center">30</td>
                  <td align="center">26</td>
                  <td align="center">32</td>
                  <td align="center">29</td>
                </tr>
                <tr>
                  <td>
                    <bold>pO</bold>
                    <sup>
                      <bold>2</bold>
                    </sup>
                    <bold>(mmHg)</bold>
                  </td>
                  <td align="center">25</td>
                  <td align="center">
                    <bold>41</bold>
                  </td>
                  <td align="center">32</td>
                  <td align="center">
                    <bold>70</bold>
                  </td>
                </tr>
                <tr>
                  <td>
                    <bold>O</bold>
                    <sup>
                      <bold>2</bold>
                    </sup>
                    <bold>sat (%)</bold>
                  </td>
                  <td align="center">52</td>
                  <td align="center">
                    <bold>83</bold>
                  </td>
                  <td align="center">65</td>
                  <td align="center">
                    <bold>95</bold>
                  </td>
                </tr>
                <tr>
                  <td>
                    <bold>HCO</bold>
                    <sup>
                      <bold>3-</bold>
                    </sup>
                    <bold>(mmol/L)</bold>
                  </td>
                  <td align="center">21</td>
                  <td align="center">19</td>
                  <td align="center">20</td>
                  <td align="center">20</td>
                </tr>
              </tbody>
            </table>
          </table-wrap>
        </p>
      </sec>
      <sec sec-type="discussion">
        <title>Discussion</title>
        <p>The prevalence of PFO in the general population is estimated to be ~25%.<sup><xref ref-type="bibr" rid="B1">1</xref></sup> In most cases, the interatrial shunt is hemodynamically trivial. However, in rare circumstances, a R-L shunt through a PFO may cause clinically significant arterial deoxygenation by mixing venous and arterial blood. These patients usually present a platypnea-orthodeoxia syndrome, a rare condition characterized by dyspnea and arterial deoxygenation induced by an upright position and typically relieved by lying supine.<sup><xref ref-type="bibr" rid="B2">2</xref></sup></p>
        <p>The occurrence of R-L interatrial shunting is usually associated with spontaneous or induced pulmonary hypertension. The occurrence of this shunt with normal pulmonary artery pressure is very uncommon, but has been described in previous case reports. This occurs by preferential blood flow streaming from the inferior vena cava into the left atrium, through the PFO, even in the absence of an interatrial pressure gradient. A prominent Eustachian valve and right chamber anatomy modification can act as contributing factors. Such a syndrome has been described in patients with mechanical conditions, causing atrial or septal deformity, such as kyphoscoliosis,<sup><xref ref-type="bibr" rid="B3">3</xref></sup> restrictive lung disease, previous pneumonectomy,<sup><xref ref-type="bibr" rid="B4">4</xref></sup> pleural effusion,<sup><xref ref-type="bibr" rid="B5">5</xref></sup> diaphragmatic paralysis and ascension,<sup><xref ref-type="bibr" rid="B6">6</xref></sup> ascending aorta aneurysm,<sup><xref ref-type="bibr" rid="B3">3</xref></sup> or post-thoracotomy.<sup><xref ref-type="bibr" rid="B7">7</xref></sup> In these cases, the anatomic relationship between the atrial septum and the inferior vena cava was changed, facilitating desaturated blood flow redirection through the PFO.</p>
        <p>The history of symptoms can be short and may have an acute onset with rapid worsening within a few days. As such, the diagnosis of an arterial deoxygenation syndrome is usually a &#8220;rule-out&#8221; diagnosis.<sup><xref ref-type="bibr" rid="B2">2</xref></sup> Common causes of acute hypoxemia must be first excluded, such as pneumonia, acute heart failure, pulmonary embolism, or other structural lung disease. In our case, transthoracic echocardiography had missed the diagnosis. The first hint came from the VQ lung scan, requested to rule out pulmonary embolism or other VQ imbalances, which revealed kidney and brain uptake of tracer, a finding that is diagnostic of a R-L shunt.<sup><xref ref-type="bibr" rid="B8">8</xref></sup> TOE confirmed the R-L shunt through the streaming of blood flow from the inferior vena cava to the PFO. Upon review of CT images, we found that the abdominal distention due to postoperative ileus had caused diaphragm elevation and cardiac deformation, which in this case was responsible for the blood streaming. After an extensive review of the literature, we found that this is the first reported case of an arterial deoxygenation syndrome due to PFO under these circumstances. Another unique feature of this case was the severe hypoxemia while lying supine, as opposed to the typical relief of deoxygenation in the supine position of patients with platypnea-orthodeoxia syndrome. This suggests that the anatomic deformation leading to blood streaming was independent of the body position.</p>
        <p>A potential limitation of the documentation of this case was that a thorough blood gas analysis in different body positions was not performed. This was due to the fact that severe hypoxemia had already been documented in decubitus, with no significant change in pulse oximetry in the sitting or standing position, so additional radial puncture seemed clinically futile at the time.</p>
      </sec>
      <sec sec-type="conclusions">
        <title>Conclusion</title>
        <p>The present case illustrates the diagnosis and successful treatment of a rare cause of hypoxemia and highlights the mechanisms causing abnormal cardiac flow and impaired oxygenation with cardiac R-L shunts, which in rare cases can occur despite normal chamber pressures.</p>
      </sec>
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    <back>
      <fn-group>
        <fn fn-type="other">
          <p>Study Association</p>
          <p>This study is not associated with any thesis or dissertation work.</p>
        </fn>
      </fn-group>
      <fn-group>
        <fn fn-type="supported-by">
          <p>Sources of Funding: There were no external funding sources for this study.</p>
        </fn>
      </fn-group>
    </back>
  </sub-article>
</article>
