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  <front>
    <journal-meta>
      <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-loc>S&#227;o Paulo, SP, Brazil</publisher-loc>
      </publisher>
    </journal-meta>
    <article-meta>
      <article-id pub-id-type="publisher-id" specific-use="scielo-v3">wGp4L7dBxg8Mb7S8Mpc53Zh</article-id>
      <article-id pub-id-type="other">00007</article-id>
      <article-id pub-id-type="publisher-id" specific-use="scielo-v2">S0066-782X2005001800007</article-id>
      <article-id pub-id-type="publisher-id" specific-use="scielo-v1">S0066-782X(05)08500507</article-id>
      <article-id pub-id-type="doi">10.1590/S0066-782X2005001800007</article-id>
      <article-categories>
        <subj-group subj-group-type="heading">
          <subject>Relatos de Caso</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title>F&#237;stula de enxerto coronariano da art&#233;ria tor&#225;cica interna esquerda para art&#233;ria pulmonar esquerda ap&#243;s cirurgia de revasculariza&#231;&#227;o mioc&#225;rdica: causa rara de isquemia mioc&#225;rdica</article-title>
      </title-group>
      <contrib-group>
        <contrib contrib-type="author">
          <name>
            <surname>Almeida J&#250;nior</surname>
            <given-names>Gustavo Luiz Gouv&#234;a de</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Jorge</surname>
            <given-names>Jos&#233; Kezen Camilo</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Neno</surname>
            <given-names>Augusto C&#233;sar de Ara&#250;jo</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Nogueira</surname>
            <given-names>Fernanda Beloni dos Santos</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Hellmuth</surname>
            <given-names>Bruno</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Lins</surname>
            <given-names>Roberto Hugo da Costa</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Vilella</surname>
            <given-names>Renato</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Carreira</surname>
            <given-names>Valdo Jos&#233;</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Thadeu</surname>
            <given-names>Ivo</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Silva</surname>
            <given-names>Jos&#233; Pedro da</given-names>
          </name>
          <xref ref-type="aff" rid="aff01"/>
        </contrib>
      </contrib-group>
      <aff id="aff01">
        <addr-line>
          <named-content content-type="city">Rio de Janeiro</named-content>
          <named-content content-type="state"/>
        </addr-line>
        <institution content-type="orgname">Casa de Sa&#250;de S&#227;o Jos&#233;</institution>
        <country country="BR">Brazil</country>
      </aff>
      <pub-date date-type="collection" publication-format="electronic">
        <month>11</month>
        <year>2005</year>
      </pub-date>
      <volume>85</volume>
      <issue>5</issue>
      <fpage>337</fpage>
      <lpage>339</lpage>
      <history>
        <date date-type="received">
          <day>04</day>
          <month>03</month>
          <year>2005</year>
        </date>
        <date date-type="accepted">
          <day>06</day>
          <month>04</month>
          <year>2005</year>
        </date>
      </history>
      <permissions>
        <license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/4.0/" xml:lang="en">
          <license-p>This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.</license-p>
        </license>
      </permissions>
      <abstract>
        <p>Descrevemos o caso de um paciente que, seis anos ap&#243;s cirurgia de revasculariza&#231;&#227;o do mioc&#225;rdio, desenvolveu dispn&#233;ia aos pequenos esfor&#231;os. Foi documentada isquemia mioc&#225;rdica por m&#233;todo de medicina nuclear e a cineangiocoronariografia mostrou todos os enxertos patentes com grande f&#237;stula da art&#233;ria tor&#225;cica interna esquerda para art&#233;ria pulmonar esquerda. O paciente foi tratado com fechamento cir&#250;rgico da f&#237;stula, tendo &#243;tima evolu&#231;&#227;o p&#243;s-operat&#243;ria.</p>
      </abstract>
      <pub-date publication-format="electronic" date-type="pub">
        <day>07</day>
        <month>12</month>
        <year>2005</year>
      </pub-date>
    </article-meta>
  </front>
  <body>
    <p>
      <bold>RELATO DE CASO</bold>
    </p>
    <p>F&#237;stula de enxerto coronariano da art&#233;ria tor&#225;cica interna esquerda para art&#233;ria pulmonar esquerda ap&#243;s cirurgia de revasculariza&#231;&#227;o mioc&#225;rdica. Causa rara de isquemia mioc&#225;rdica</p>
    <p>
      <bold>Gustavo Luiz Gouv&#234;a de Almeida J&#250;nior; Jos&#233; Kezen Camilo Jorge; Augusto C&#233;sar de Ara&#250;jo Neno; Fernanda Beloni dos Santos Nogueira; Bruno Hellmuth; Roberto Hugo da Costa Lins; Renato Vilella; Valdo Jos&#233; Carreira; Ivo Thadeu; Jos&#233; Pedro da Silva</bold>
    </p>
    <p>Casa de Sa&#250;de S&#227;o Jos&#233; - Rio de Janeiro, RJ</p>
    <p>
      <xref ref-type="corresp" rid="back10">Endere&#231;o para correspond&#234;ncia</xref>
    </p>
    <p>
      <bold>RESUMO</bold>
    </p>
    <p>Descrevemos o caso de um paciente que, seis anos ap&#243;s cirurgia de revasculariza&#231;&#227;o do mioc&#225;rdio, desenvolveu dispn&#233;ia aos pequenos esfor&#231;os. Foi documentada isquemia mioc&#225;rdica por m&#233;todo de medicina nuclear e a cineangiocoronariografia mostrou todos os enxertos patentes com grande f&#237;stula da art&#233;ria tor&#225;cica interna esquerda para art&#233;ria pulmonar esquerda. O paciente foi tratado com fechamento cir&#250;rgico da f&#237;stula, tendo &#243;tima evolu&#231;&#227;o p&#243;s-operat&#243;ria.</p>
    <p>A presen&#231;a de f&#237;stula da art&#233;ria tor&#225;cica interna esquerda para art&#233;ria pulmonar esquerda constitui complica&#231;&#227;o extremamente rara ap&#243;s cirurgia de revasculariza&#231;&#227;o mioc&#225;rdica. Sua presen&#231;a pode levar a recorr&#234;ncia de angina, dispn&#233;ia, insufici&#234;ncia card&#237;aca, endocardite, entre outros. Deve ser pesquisada quando nenhuma causa clara para o aparecimento desses sintomas ocorrer ap&#243;s revasculariza&#231;&#227;o mioc&#225;rdica. Seu diagn&#243;stico &#233; feito atrav&#233;s da cineangiocoronariografia e o tratamento, na maioria dos casos, &#233; o fechamento cir&#250;rgico ou percut&#226;neo da f&#237;stula.</p>
    <p>
      <bold>RELATO DO CASO</bold>
    </p>
    <p>Paciente de 73 anos de idade, masculino, branco, com hist&#243;rico de hipertens&#227;o arterial sist&#234;mica e revasculariza&#231;&#227;o mioc&#225;rdica h&#225; seis anos, com coloca&#231;&#227;o dos seguintes enxertos: art&#233;ria tor&#225;cica interna (mam&#225;ria) esquerda para art&#233;ria descendente anterior, safena seq&#252;encial para 1&#170; e 2&#170; art&#233;rias marginais esquerdas e safena para 1&#170; diagonal. O paciente procurou atendimento por estar apresentando astenia e dispn&#233;ia progressiva aos esfor&#231;os, que vem piorando nos &#250;ltimos meses, chegando aos pequenos esfor&#231;os. A medica&#231;&#227;o regular que vinha fazendo uso era captopril 150 mg/dia, hidroclorotiazida 25 mg/dia e &#225;cido acetilsalic&#237;lico 200 mg/dia. O exame f&#237;sico cardiovascular foi normal, exceto por presen&#231;a de 4&#170; bulha. Os pulm&#245;es estavam limpos. O eletrocardiograma mostrava bloqueio &#225;trio-ventricular de 1&#186; grau e hemibloqueio anterior esquerdo. O ecocardiograma revelou apenas hipertrofia ventricular esquerda e d&#233;ficit de relaxamento do ventr&#237;culo esquerdo. Foi optado, ent&#227;o, por investiga&#231;&#227;o de isquemia mioc&#225;rdica em atividade, por meio de cintilografia mioc&#225;rdica de perfus&#227;o com esfor&#231;o/repouso. A cintilografia (<xref ref-type="fig" rid="fig1">fig. 1</xref>) mostrou hipocapta&#231;&#227;o do radiof&#225;rmaco (Tc-99 sestamibi) no territ&#243;rio &#226;ntero-septal no esfor&#231;o, com recupera&#231;&#227;o no repouso, compat&#237;vel com isquemia nesse territ&#243;rio. Realizado, ent&#227;o, estudo cineangiocoronariogr&#225;fico com ventriculografia esquerda, que mostrou pat&#234;ncia de todos os enxertos e obstru&#231;&#227;o de 70% na coron&#225;ria direita. Entretanto, foi observada grande f&#237;stula saindo da por&#231;&#227;o inicial da mam&#225;ria esquerda, drenando para art&#233;ria pulmonar esquerda (<xref ref-type="fig" rid="f2">figs 2</xref> e <xref ref-type="fig" rid="f3">3</xref>). Essa f&#237;stula provocava importante roubo de fluxo da art&#233;ria descendente anterior, sendo considerado o respons&#225;vel pela isquemia no territ&#243;rio &#226;ntero-septal. O paciente foi, ent&#227;o, submetido &#224; ligadura cir&#250;rgica da f&#237;stula e recebeu alta hospitalar. No seguimento de um ano, o paciente apresentava-se livre dos sintomas relacionados com sua interna&#231;&#227;o.</p>
    <p content-type="asset">
      <fig id="f2">
        <graphic xlink:href="1678-4170-abc-85-05-0337-00007-gf01.jpg"/></fig>
    </p>
    <p content-type="asset">
      <fig id="f3">
        <graphic xlink:href="1678-4170-abc-85-05-0337-00007-gf02.jpg"/></fig>
    </p>
    <fig id="fig1">
      <graphic xlink:href="1678-4170-abc-85-05-0337-00007-gf03.jpg"/></fig>
    <p>
      <bold>DISCUSS&#195;O</bold>
    </p>
    <p>A presen&#231;a de f&#237;stulas envolvendo a art&#233;ria tor&#225;cica interna &#233; rara. Tem m&#250;ltiplas causas e, freq&#252;entemente, diagn&#243;stico dif&#237;cil, devido &#224; variabilidade na apresenta&#231;&#227;o cl&#237;nica. Pode ter evolu&#231;&#227;o de potencial gravidade e mesmo fatal. Os fatores predisponentes para o aparecimento da f&#237;stula s&#227;o a liga&#231;&#227;o incompleta dos ramos intercostais da art&#233;ria tor&#225;cica interna, les&#227;o do par&#234;nquima pulmonar, infec&#231;&#227;o e eletrocoagula&#231;&#227;o, ao inv&#233;s de ligadura dos ramos intercostais, entre outros<sup>1,2</sup>. O aparecimento de f&#237;stula da art&#233;ria tor&#225;cica interna para art&#233;ria pulmonar &#233; uma complica&#231;&#227;o rara ap&#243;s cirurgia de revasculariza&#231;&#227;o mioc&#225;rdica. Na literatura m&#233;dica, pouco mais de 20 casos foram relatados<sup>3</sup>. Os sintomas mais freq&#252;entes s&#227;o aqueles relacionados ao aparecimento de isquemia mioc&#225;rdica, como angina do peito<sup>4</sup> e dispn&#233;ia, al&#233;m de, ocasionalmente, poder ocorrer insufici&#234;ncia card&#237;aca, endarterite ou ser ouvido sopro cont&#237;nuo na borda esternal esquerda<sup>5</sup>. Com o progressivo desenvolvimento da f&#237;stula, ocorre roubo de fluxo coronariano para a art&#233;ria pulmonar e, conseq&#252;entemente, isquemia mioc&#225;rdica. No presente caso, documentamos a presen&#231;a de isquemia mioc&#225;rdica por meio da cintilografia mioc&#225;rdica e o estudo anat&#244;mico coronariano n&#227;o mostrou outra justificativa para a isquemia que n&#227;o fosse a presen&#231;a da f&#237;stula. A &#250;nica obstru&#231;&#227;o coronariana presente (coron&#225;ria direita) foi quantificada como moderada e n&#227;o correspondia ao territ&#243;rio da isquemia pelo estudo cintilogr&#225;fico.</p>
    <p>Como o paciente se apresentava sintom&#225;tico, com piora progressiva da dispn&#233;ia aos esfor&#231;os e o estudo de medicina nuclear confirmou a presen&#231;a de isquemia, foi indicado fechamento da f&#237;stula. Apesar da possibilidade de fechamento via percut&#226;nea, atrav&#233;s de <italic>stents</italic> recobertos ou de devices tipo Amplatzer e <italic>coils</italic>, apenas recentemente essas estrat&#233;gias t&#234;m sido relatadas<sup>3,6</sup> e em centros isolados. Em fun&#231;&#227;o disso e da ampla experi&#234;ncia da equipe cir&#250;rgica, foi realizado o fechamento da f&#237;stula atrav&#233;s de minitoracotomia, com excelente resultado e r&#225;pida recupera&#231;&#227;o p&#243;s-operat&#243;ria.</p>
    <p>A presen&#231;a de recorr&#234;ncia de angina ou equivalente ap&#243;s revasculariza&#231;&#227;o mioc&#225;rdica imp&#245;e diagn&#243;stico diferencial com f&#237;stula da mam&#225;ria, se uma causa clara para os sintomas n&#227;o for identificada, uma vez que constitui causa potencialmente grave e que requer tratamento espec&#237;fico. O diagn&#243;stico definitivo &#233; feito atrav&#233;s da coronariografia com inje&#231;&#227;o seletiva na art&#233;ria tor&#225;cica interna e o tratamento padr&#227;o &#233; a ligadura cir&#250;rgica da f&#237;stula. Como alternativa, pode ser realizado tratamento atrav&#233;s de t&#233;cnicas percut&#226;neas, caso equipe de hemodin&#226;mica esteja familiarizada com esse tipo de procedimento.</p>
    <p>
      <fn id="back10" fn-type="corresp">
        <label>
          <bold>Endere&#231;o para correspond&#234;ncia</bold>
        </label>
        <p> Gustavo Luiz Gouv&#234;a de Almeida J&#250;nior </p>
        <p> Rua Alfredo Ceschiatti, 100 Bl. 01/1206 - Barra da Tijuca </p>
        <p> 22775-045 - Rio de Janeiro, RJ </p>
        <p> E-mail: </p>
        <p>
          <email xlink:href="gustavogouvea@globo.com">gustavogouvea@globo.com</email>
        </p>
      </fn>
    </p>
    <p>Recebido em 04/03/05 </p>
    <p> Aceito em 06/04/05</p>
  </body>
  <back>
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  </back>
  <sub-article article-type="translation" id="TRen" xml:lang="en">
    <front-stub>
      <article-categories>
        <subj-group subj-group-type="heading">
          <subject>Case Reports</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title xml:lang="en">Left internal thoracic artery to left pulmonary artery fistula after coronary artery bypass graft surgery: a rare cause of myocardial ischemia</article-title>
      </title-group>
      <abstract xml:lang="en">
        <p>We report a patient who developed dyspnea on mild exertion six years after coronary artery bypass graft surgery (CABG). Myocardial ischemia was documented by radionuclide imaging, and coronary angiography showed patency of all grafts and a large fistula between the left internal thoracic artery (LITA) and the left pulmonary artery (LPA). The patient was submitted to surgical closure of the fistula and made an excellent recovery.</p>
      </abstract>
    </front-stub>
    <body>
      <p>
        <bold>CASE REPORT</bold>
      </p>
      <p>Left internal thoracic artery to left pulmonary artery fistula after coronary artery bypass graft surgery. A rare cause of myocardial ischemia</p>
      <p>
        <bold>Gustavo Luiz Gouv&#234;a de Almeida J&#250;nior; Jos&#233; Kezen Camilo Jorge; Augusto C&#233;sar de Ara&#250;jo Neno; Fernanda Beloni dos Santos Nogueira; Bruno Hellmuth; Roberto Hugo da Costa Lins; Renato Vilella; Valdo Jos&#233; Carreira; Ivo Thadeu; Jos&#233; Pedro da Silva</bold>
      </p>
      <p>Casa de Sa&#250;de S&#227;o Jos&#233; - Rio de Janeiro, RJ - Brazil</p>
      <p>
        <xref ref-type="corresp" rid="back10-body2">Mailing address</xref>
      </p>
      <p>
        <bold>ABSTRACT</bold>
      </p>
      <p>We report a patient who developed dyspnea on mild exertion six years after coronary artery bypass graft surgery (CABG). Myocardial ischemia was documented by radionuclide imaging, and coronary angiography showed patency of all grafts and a large fistula between the left internal thoracic artery (LITA) and the left pulmonary artery (LPA). The patient was submitted to surgical closure of the fistula and made an excellent recovery.</p>
      <p>Left internal thoracic artery fistula draining to left pulmonary artery is an extremely rare complication following myocardial revascularization. It may cause recurrent angina, dyspnea, heart failure, endocarditis, among other conditions. It should always be considered in the absence of a clear cause for the onset of these symptoms after myocardial revascularization. The diagnosis is made by coronary angiography, and most patients are treated by surgical or percutaneous closure of the fistula.</p>
      <p>
        <bold>CASE REPORT</bold>
      </p>
      <p>A 73-year-old white male patient with a history of systemic arterial hypertension and coronary artery bypass grafting six years ago using the following grafts: left internal thoracic (mammary) artery to anterior descending artery, sequential saphenous vein to the 1<sup>st</sup> and 2<sup>nd</sup> left marginal arteries and saphenous vein to the 1<sup>st</sup> diagonal artery. The patient presented with asthenia and progressive exertional dyspnea, which has been getting worse over the last few months even on mild exertion. He had been taking captopril 150 mg/d, hydrochlorothiazide 25 mg/d, and acetylsalicylic acid 200 mg/d. The cardiovascular physical examination was normal, except for a fourth heart sound (S4). His lungs were found to be clean. First-degree atrioventricular block and left anterior hemiblock was seen on ECG. The echocardiogram revealed only left ventricular hypertrophy and diastolic disfunction. Myocardial ischemia investigation was performed through stress/rest myocardial perfusion scintigraphy. The scan (<xref ref-type="fig" rid="fig1-body2">fig.1</xref>) showed low uptake of the radiopharmaceutical (Tc-99 sestamibi) in the anteroseptal region on stress, which returned to normal at rest, a finding consistent with anteroseptal ischemia. Coronary angiography and left ventriculography were thus performed, showing patency of all grafts and a 70% obstruction of the right coronary artery. However, a large fistula arising at the initial portion of the left mammary artery draining to the left pulmonary artery was detected (<xref ref-type="fig" rid="en_26928f2-body2">fig. 2</xref> e <xref ref-type="fig" rid="en_26928f3-body2">3</xref>). This fistula resulted in a significant steal of flow from the anterior descending artery, thought to be the cause of the anteroseptal ischemia. A surgical ligation of the fistula was performed, and the patient was discharged from the hospital. During the one-year follow-up period, the patient was free of the symptoms which led to his hospitalization.</p>
      <p content-type="asset">
        <fig id="en_26928f2-body2">
          <graphic xlink:href="1678-4170-abc-85-05-0337-00007-gf01-en.jpg"/></fig>
      </p>
      <p content-type="asset">
        <fig id="en_26928f3-body2">
          <graphic xlink:href="1678-4170-abc-85-05-0337-00007-gf02-en.jpg"/></fig>
      </p>
      <fig id="fig1-body2">
        <graphic xlink:href="1678-4170-abc-85-05-0337-00007-gf03-en.jpg"/></fig>
      <p>
        <bold>DISCUSSION</bold>
      </p>
      <p>Fistulas involving internal thoracic artery are rare. They have multiple causes, and diagnosis is often difficult to make due to the variable clinical presentation. They may evolve into a severe condition or even cause death. The predisposing factors for the onset of fistula include incomplete ligation of the intercostal branches of the internal thoracic artery, injury to the pulmonary parenchyma, infection, and electrocoagulation, instead of ligation of the intercostal branches, among others <sup>1,2</sup>. Fistula of the internal thoracic artery to pulmonary artery is a rare complication following CABG. Around 20 or more cases were reported in the medical literature <sup>3</sup>. The most common symptoms are those related to the onset of myocardial ischemia, such as angina pectoris and dyspnea,<sup>4</sup> and occasionally heart failure, endarteritis or a continuous murmur heard at the left sternal border.<sup>5</sup> As the fistula continues to develop, a coronary steal of flow to the pulmonary artery occurs, resulting in myocardial ischemia. In the present case, the myocardial ischemia was documented by myocardial scintigraphy, and the anatomical study of the coronary vessels showed no other explanation for the ischemia but the presence of the fistula. The only existent coronary obstruction (right coronary artery) was evaluated as moderate, and did not correspond to the ischemia territory shown on the scintigraphic study.</p>
      <p>As the patient was symptomatic with exertional dyspnea growing worse, and nuclear medicine techniques attested to the presence of ischemia, closure of the fistula was indicated. Despite the possibility of percutaneous closure with covered stents or devices like Amplatzer and coils, only recently these strategies have been reported,<sup>3,6</sup> and by a few medical centers. Considering this and the large experience of the surgery team, the closure of the fistula through minithoracotomy was successfully performed, and the patient had a rapid postoperative recovery.</p>
      <p>The presence of recurrent angina or an equivalent condition after myocardial revascularization requires differential diagnosis from mammary fistula, in case a clear cause for the symptoms is not found, because it is potentially serious and requires specific treatment. The definitive diagnosis is made angiographically with selective injection in the internal thoracic artery and surgical ligation being the standard treatment for fistula. Alternatively, it can be done through percutaneous techniques, providing the hemodynamic team is familiar with the procedure.</p>
      <p>
        <bold>REFERENCES</bold>
      </p>
      <p>
        <fn id="back10-body2" fn-type="corresp">
          <label>
            <bold>Mailing address</bold>
          </label>
          <p> Gustavo Luiz Gouv&#234;a de Almeida J&#250;nior</p>
          <p> Rua Alfredo Ceschiatti, 100 Bl. 01/1206</p>
          <p> Barra da Tijuca</p>
          <p> 22775-045 - Rio de Janeiro, RJ - Brazil</p>
          <p> E-mail: </p>
          <p>
            <email xlink:href="gustavogouvea@globo.com">gustavogouvea@globo.com</email>
          </p>
        </fn>
      </p>
      <p>Received on 03/04/05</p>
      <p> Accepted on 04/06/05</p>
    </body>
  </sub-article>
</article>
