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<article xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" article-type="case-report" 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 pub-id-type="publisher-id" specific-use="scielo-v3">4bgGG7g8nZbXTvzFYPwZ5Xq</article-id>
      <article-id pub-id-type="publisher-id" specific-use="scielo-v2">S0066-782X2020000500034</article-id>
      <article-id pub-id-type="publisher-id">abc.20190295</article-id>
      <article-id pub-id-type="doi">10.36660/abc.20190295</article-id>
      <article-categories>
        <subj-group subj-group-type="heading">
          <subject>Relato de Caso</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title>S&#237;ndrome do Bloqueio de Ramo Esquerdo Doloroso em Paciente Encaminhada para Estudo Eletrofisiol&#243;gico: Um Relato de Caso</article-title>
      </title-group>
      <contrib-group>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0002-3835-6067</contrib-id>
          <name>
            <surname>Alencar</surname>
            <given-names>Jos&#233; Nunes de</given-names>
            <suffix>Neto</suffix>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Sakai</surname>
            <given-names>Marcel Henrique</given-names>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Moraes</surname>
            <given-names>Saulo Rodrigo Ramalho de</given-names>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Frota</surname>
            <given-names>Elano Sousa da</given-names>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0001-7290-9158</contrib-id>
          <name>
            <surname>Cirenza</surname>
            <given-names>Claudio</given-names>
          </name>
          <xref ref-type="aff" rid="aff1">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Paola</surname>
            <given-names>Angelo Amato Vincenzo de</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">Universidade Federal de S&#227;o Paulo</institution>
        <institution content-type="orgdiv1">Escola Paulista de Medicina</institution>
        <addr-line>
          <named-content content-type="city">S&#227;o Paulo</named-content>
          <named-content content-type="state">SP</named-content>
        </addr-line>
        <country country="BR">Brasil</country>
        <institution content-type="original">Universidade Federal de S&#227;o Paulo Escola Paulista de Medicina, S&#227;o Paulo, SP - Brasil</institution>
      </aff>
      <author-notes>
        <corresp id="c01"> Correspond&#234;ncia: Jos&#233; Nunes de Alencar Neto &#8226; Universidade Federal de S&#227;o Paulo Escola Paulista de Medicina - Rua Napole&#227;o de Barros, 715. CEP 04023-062, S&#227;o Paulo, SP &#8211; Brasil E-mail: <email>josenunesalencar@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: Alencar Neto JN, Cirenza C, Paola AAV; Obten&#231;&#227;o de dados: Alencar Neto JN, Sakai MH, Moraes SRR, Frota ES; Reda&#231;&#227;o do manuscrito: Alencar Neto JN, Sakai MH, Moraes SRR, Frota ES, Paola AAV; Revis&#227;o cr&#237;tica do manuscrito quanto ao conte&#250;do intelectual importante: Cirenza C, Paola AAV.</p>
        </fn>
        <fn fn-type="conflict">
          <p>Potencial conflito de interesses</p>
          <p>Declaro n&#227;o haver conflito de interesses pertinentes.</p>
        </fn>
      </author-notes>
      <pub-date date-type="pub" publication-format="electronic">
        <day>18</day>
        <month>05</month>
        <year>2020</year>
      </pub-date>
      <pub-date date-type="collection" publication-format="electronic">
        <month>04</month>
        <year>2020</year>
      </pub-date>
      <volume>114</volume>
      <issue>4 suppl 1</issue>
      <fpage>34</fpage>
      <lpage>37</lpage>
      <history>
        <date date-type="received">
          <day>30</day>
          <month>08</month>
          <year>2018</year>
        </date>
        <date date-type="rev-recd">
          <day>03</day>
          <month>05</month>
          <year>2019</year>
        </date>
        <date date-type="accepted">
          <day>18</day>
          <month>08</month>
          <year>2019</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 Non-Commercial License, which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. </license-p>
        </license>
      </permissions>
      <kwd-group xml:lang="pt">
        <kwd>Bloqueio Card&#237;aco</kwd>
        <kwd>Dor no Peito</kwd>
        <kwd>Doen&#231;a Arterial Coronariana/fisiopatologia</kwd>
        <kwd>Eletrofisiologia Card&#237;aca</kwd>
        <kwd>Eletrocardiografia</kwd>
        <kwd>Ecocardiografia</kwd>
      </kwd-group>
      <counts>
        <fig-count count="6"/>
        <table-count count="2"/>
        <equation-count count="0"/>
        <ref-count count="10"/>
        <page-count count="4"/>
      </counts>
    </article-meta>
  </front>
  <body>
    <sec sec-type="intro">
      <title>Introdu&#231;&#227;o</title>
      <p>O desenvolvimento de dor precordial associada ao bloqueio de ramo esquerdo (BRE) intermitente na aus&#234;ncia de doen&#231;a arterial coronariana tem sido descrito na literatura como s&#237;ndrome do bloqueio de ramo esquerdo doloroso. O mecanismo respons&#225;vel pela dor precordial &#233; desconhecido, mas a principal hip&#243;tese atualmente est&#225; relacionada &#224; dissincronia card&#237;aca aguda.</p>
      <p>Nessa s&#237;ndrome, o BRE ocorre quando a dura&#231;&#227;o do ciclo &#233; igual ou inferior ao per&#237;odo refrat&#225;rio do ramo esquerdo, principalmente durante o esfor&#231;o f&#237;sico. A dor tor&#225;cica na s&#237;ndrome do BRE doloroso pode variar entre um leve desconforto a uma condi&#231;&#227;o incapacitante.</p>
      <p>Esse relato descreve uma paciente com BRE frequ&#234;ncia-dependente t&#237;pico associado com dor tor&#225;cica, encaminhada ao estudo eletrofisiol&#243;gico (EEF) sem evid&#234;ncias de arritmias.</p>
    </sec>
    <sec>
      <title>Relato de caso</title>
      <p>Paciente do sexo feminino, 41 anos, com hist&#243;rico de hipertens&#227;o controlada e dois anos de palpita&#231;&#245;es associadas com dor tor&#225;cica desencadeada por esfor&#231;o m&#237;nimo durante atividades cotidianas, que persistia por at&#233; 2 horas. A dor tor&#225;cica foi descrita como uma sensa&#231;&#227;o de press&#227;o, que irradiava para o bra&#231;o esquerdo, associada a n&#225;usea e dispneia. Os epis&#243;dios foram caracterizados por in&#237;cio s&#250;bito, sem pr&#243;dromos, com melhora espont&#226;nea. Inicialmente, a paciente foi tratada com atenolol 25 mg (duas vezes ao dia), com al&#237;vio parcial dos sintomas. N&#227;o havia hist&#243;rico familiar de s&#237;ncope inexplicada ou morte card&#237;aca s&#250;bita. O exame f&#237;sico foi normal. O ECG de 12 deriva&#231;&#245;es durante a crise revelou taquicardia de complexo alargado com bloqueio de ramo esquerdo completo (BRE), eixo inferior e onda P compat&#237;vel com ritmo sinusal. Mesmo assim, a paciente foi referia ao EEF, que n&#227;o mostrou substratos arritmog&#234;nicos.</p>
      <p>Entretanto, no in&#237;cio da estimula&#231;&#227;o atrial cont&#237;nua a 600 ms, foi observado um BRE frequ&#234;ncia dependente. Imediatamente ap&#243;s o BRE, a paciente, que n&#227;o havia sido mantida sedada, come&#231;ou a se queixar dos mesmos sintomas j&#225; descritos. O BRE persistiu por alguns minutos e desapareceu espontaneamente, concomitantemente com o al&#237;vio da dor. O ECG &#233; apresentado na <xref ref-type="fig" rid="f01">Figura 1</xref> . Trata-se de um BRE de 3&#186; grau t&#237;pico, com dura&#231;&#227;o do complexo QRS de 138 ms, eixo superior e onda P sinusal.</p>
      <p>
        <fig id="f01">
          <label>Figura 1</label>
          <caption>
            <title>
              <italic><bold>&#8211;</bold> ECG de 12 deriva&#231;&#245;es demonstrando o in&#237;cio da dor imediatamente ap&#243;s o bloqueio de ramo esquerdo.</italic>
            </title>
          </caption>
          <graphic xlink:href="1678-4170-abc-114-4suppl1-0034-90295-gf01.jpg"/></fig>
      </p>
      <p>O ECG basal de 12 deriva&#231;&#245;es estava normal ( <xref ref-type="fig" rid="f02">Figura 2</xref> ). O monitoramento por Holter de 24 horas revelou que a FC basal variou entre 56 e 116 bpm durante as atividades cotidianas, sem evid&#234;ncia de BRE. Tanto a ecocardiografia transtor&#225;cica quanto a resson&#226;ncia magn&#233;tica card&#237;aca mostraram fun&#231;&#227;o sist&#243;lica normal sem doen&#231;a mioc&#225;rdica ou valvar. Todas as c&#226;maras card&#237;acas estavam com tamanho normal. Um teste de esfor&#231;o revelou o desenvolvimento de bloqueio de ramo esquerdo associado com a dor tor&#225;cica. A angiotomografia descartou doen&#231;a arterial coronariana e defeitos de perfus&#227;o do mioc&#225;rdio com diripidamol. Atualmente, a paciente est&#225; recebendo atenolol 50 mg (duas vezes ao dia) e n&#227;o houve recorr&#234;ncias de palpita&#231;&#245;es ou dor precordial no acompanhamento de seis meses.</p>
      <p>
        <fig id="f02">
          <label>Figura 2</label>
          <caption>
            <title>
              <italic><bold>&#8211;</bold> ECG basal de 12 deriva&#231;&#245;es normal.</italic>
            </title>
          </caption>
          <graphic xlink:href="1678-4170-abc-114-4suppl1-0034-90295-gf02.jpg"/></fig>
      </p>
    </sec>
    <sec sec-type="discussion">
      <title>Discuss&#227;o</title>
      <p>Em 1946, foi publicado o primeiro relato de bloqueio de ramo esquerdo intermitente relacionado a esfor&#231;os. O paciente apresentava palpita&#231;&#245;es e sensa&#231;&#227;o de dor no prec&#243;rdio durante as crises. Entretanto, a angiocoronariografia n&#227;o foi realizada devido &#224; tecnologia dispon&#237;vel naquela &#233;poca.<sup><xref ref-type="bibr" rid="B1">1</xref></sup> Em 1976, Vieweg et al.,<sup><xref ref-type="bibr" rid="B2">2</xref></sup> relataram o primeiro caso de bloqueio de ramo esquerdo associado com angina de esfor&#231;o, com evid&#234;ncias angiogr&#225;ficas de art&#233;rias coron&#225;rias normais. Embora tenha sido feito um diagn&#243;stico de angina, foram observadas caracter&#237;sticas at&#237;picas: in&#237;cio e fim abruptos da dor, concomitantemente ao BRE e ap&#243;s o seu desaparecimento, respectivamente.<sup><xref ref-type="bibr" rid="B2">2</xref></sup> Em 1982, Virtanen et al.,<sup><xref ref-type="bibr" rid="B3">3</xref></sup> conduziram um estudo com 7 pacientes portadores de bloqueio de ramo esquerdo recente e dor precordial durante o teste de esfor&#231;o, todos com angiocoronariografias normais. Nesse estudo, foi avaliado o padr&#227;o da dor apresentada pelos pacientes. Em todos os casos, a dor foi considerada at&#237;pica por conta do in&#237;cio e fim abruptos.<sup><xref ref-type="bibr" rid="B3">3</xref></sup> Posteriormente, novos casos foram relatados, e a essa condi&#231;&#227;o deu-se o nome de s&#237;ndrome do bloqueio de ramo esquerdo doloroso.</p>
      <p>Os mecanismos da s&#237;ndrome do BRE doloroso s&#227;o pouco claros. A possibilidade de isquemia de demanda resultante de les&#245;es ou espasmos coron&#225;rios foi inicialmente considerada uma poss&#237;vel causa para essa s&#237;ndrome, mas logo essa suposi&#231;&#227;o se mostrou incorreta. O in&#237;cio/fim imediatos da dor s&#227;o incompat&#237;veis com isquemia.<sup><xref ref-type="bibr" rid="B4">4</xref></sup> A nitroglicerina se mostrou ineficaz<sup><xref ref-type="bibr" rid="B2">2</xref></sup> e, &#224;s vezes, induziu o BRE devido &#224; taquicardia. Muitas vezes, o resultado do exame de imagem nuclear era negativo e o vasoespasmo tamb&#233;m havia sido descartado.<sup><xref ref-type="bibr" rid="B5">5</xref> , <xref ref-type="bibr" rid="B6">6</xref></sup></p>
      <p>A melhor teoria at&#233; agora &#233; a proposta por Virtanen et al.,<sup><xref ref-type="bibr" rid="B3">3</xref></sup> que, por meio da avalia&#231;&#227;o de ventriculografias, especulou que a dor poderia ser induzida pelo movimento sist&#243;lico anormal do septo. A presen&#231;a do eixo inferior em uma s&#233;rie de casos de maneira uniforme fez com que os autores presumissem a exist&#234;ncia de um padr&#227;o de contratilidade espec&#237;fico. Shvlikin et al.,<sup><xref ref-type="bibr" rid="B7">7</xref></sup> propuseram crit&#233;rios para o diagn&#243;stico da s&#237;ndrome do BRE doloroso ( <xref ref-type="table" rid="t1">Tabela 1</xref> ).<sup><xref ref-type="bibr" rid="B7">7</xref></sup></p>
      <p>
        <table-wrap id="t1">
          <label>Tabela 1</label>
          <caption>
            <title>&#8211; Crit&#233;rios para a s&#237;ndrome do BRE doloroso</title>
          </caption>
          <table frame="hsides" rules="groups">
            <colgroup>
              <col/>
            </colgroup>
            <tbody>
              <tr>
                <td align="center">In&#237;cio abrupto de dor tor&#225;cica com desenvolvimento do BRE</td>
              </tr>
              <tr>
                <td align="center">Resolu&#231;&#227;o concomitante dos sintomas com resolu&#231;&#227;o do BRE (ocasionalmente ausente)</td>
              </tr>
              <tr>
                <td align="center">ECG de 12 deriva&#231;&#245;es normais antes e ap&#243;s o BRE</td>
              </tr>
              <tr>
                <td align="center">Aus&#234;ncia de isquemia mioc&#225;rdica durante a prova de estresse funcional</td>
              </tr>
              <tr>
                <td align="center">Fun&#231;&#227;o ventricular esquerda normal e aus&#234;ncia de outras condi&#231;&#245;es que possam explicar os sintomas</td>
              </tr>
              <tr>
                <td align="center">Rela&#231;&#227;o S/T &lt;1,8 em deriva&#231;&#245;es precordiais e eixo inferior</td>
              </tr>
            </tbody>
          </table>
          <table-wrap-foot>
            <fn id="TFN1">
              <p>
                <italic>Crit&#233;rios propostos para o diagn&#243;stico da s&#237;ndrome do BRE doloroso. Adaptado de Shvilkin.<sup>7</sup></italic>
              </p>
            </fn>
          </table-wrap-foot>
        </table-wrap>
      </p>
      <p>De forma semelhante &#224; onda T da mem&#243;ria card&#237;aca de pacientes com marcapasso, o BRE cr&#244;nica apresenta ondas T de menor amplitude do que a BRE aguda. Em estudo prospectivo, uma rela&#231;&#227;o S/T &lt; 2,5 em deriva&#231;&#245;es precordiais se mostrou eficaz (100% sensibilidade e 89% especificidade) para distinguir entre o BRE de in&#237;cio recente ou cr&#244;nico,<sup><xref ref-type="bibr" rid="B8">8</xref></sup> um dos itens dos crit&#233;rios propostos na <xref ref-type="table" rid="t1">Tabela 1</xref> .</p>
      <p>A paciente a qual se refere este artigo foi encaminhada ao EEF por conta de uma hip&#243;tese equ&#237;voca de taquicardia supraventricular com aberr&#226;ncia. Durante o estudo, com a estimula&#231;&#227;o atrial cont&#237;nua, tivemos a oportunidade de registrar o momento exato do bloqueio de ramo esquerdo e a imediata queixa sobre a mesma dor previamente referida pela paciente como sendo cr&#244;nica.</p>
      <p>Ao tra&#231;armos uma compara&#231;&#227;o com os crit&#233;rios propostos por Schvilkin et al.,<sup><xref ref-type="bibr" rid="B8">8</xref></sup> verificamos que nosso caso se encaixa em todos os crit&#233;rios, com a exce&#231;&#227;o de um: o &#8220;crit&#233;rio do eixo inferior&#8221;. Entretanto, outras publica&#231;&#245;es tamb&#233;m mostraram um complexo QRS superior.<sup><xref ref-type="bibr" rid="B9">9</xref> , <xref ref-type="bibr" rid="B10">10</xref></sup> A rela&#231;&#227;o S/T foi igual a 1,33 em V2 ( <xref ref-type="fig" rid="f03">Figura 3</xref> ), compat&#237;vel com um BRE de in&#237;cio agudo. A paciente apresentou in&#237;cio abrupto de dor, conforme registrado pelos membros da nossa equipe no laborat&#243;rio de eletrofisiologia. O desaparecimento dos sintomas ocorreu imediatamente ap&#243;s o desaparecimento do BRE. O ECG basal de 12 deriva&#231;&#245;es estava normal. Um teste de esfor&#231;o descartou a isquemia mioc&#225;rdica e a angiotomografia revelou art&#233;rias coron&#225;rias normais. Tanto a ecocardiografia quanto a resson&#226;ncia card&#237;aca estavam normais, com exce&#231;&#227;o das causas secund&#225;rias da angina.</p>
      <p>
        <fig id="f03">
          <label>Figura 3</label>
          <caption>
            <title>
              <italic><bold>&#8211;</bold> Rela&#231;&#227;o S/T &lt; 1,8 em V2.</italic>
            </title>
          </caption>
          <graphic xlink:href="1678-4170-abc-114-4suppl1-0034-90295-gf03.jpg"/></fig>
      </p>
    </sec>
    <sec sec-type="conclusions">
      <title>Conclus&#227;o</title>
      <p>Relatamos um caso de BRE doloroso de uma paciente encaminhada ao EEF. O in&#237;cio abrupto da dor assim que o bloqueio do ramo esquerdo ocorre &#233; incompat&#237;vel com a isquemia. Al&#233;m disso, a paciente foi submetida a exames que descartaram o comprometimento coron&#225;rio e mioc&#225;rdico. A melhor hip&#243;tese para a fisiopatologia dessa s&#237;ndrome &#233; a dissincronia dolorosa do cora&#231;&#227;o resultante do BRE de in&#237;cio agudo. At&#233; onde sabemos, este &#233; o primeiro relato de caso sobre essa s&#237;ndrome em uma revista m&#233;dica brasileira.</p>
    </sec>
  </body>
  <back>
    <ref-list>
      <title>Refer&#234;ncias</title>
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        <label>1</label>
        <mixed-citation>1. Eichert H. Transient bundle branch block associated with tachycardia. Am Heart J. United States; 1946 Apr;31:511&#8211;8.</mixed-citation>
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            <name>
              <surname>Heikkila</surname>
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          <year>1982</year>
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        <p>Vincula&#231;&#227;o acad&#234;mica</p>
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      </fn>
      <fn fn-type="other">
        <p>Aprova&#231;&#227;o &#233;tica e consentimento informado</p>
        <p>Este artigo n&#227;o cont&#233;m estudos com humanos ou animais realizados por nenhum dos autores.</p>
      </fn>
    </fn-group>
    <fn-group>
      <fn fn-type="supported-by">
        <p>Fontes de financiamento</p>
        <p>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>Case Report</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title>Painful Left Bundle Branch Block Syndrome in a Patient Referred to Electrophysiologic Study: A Case Report</article-title>
      </title-group>
      <contrib-group>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0002-3835-6067</contrib-id>
          <name>
            <surname>Alencar</surname>
            <given-names>Jos&#233; Nunes de</given-names>
            <suffix>Neto</suffix>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Sakai</surname>
            <given-names>Marcel Henrique</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Moraes</surname>
            <given-names>Saulo Rodrigo Ramalho de</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Frota</surname>
            <given-names>Elano Sousa da</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <contrib-id contrib-id-type="orcid">0000-0001-7290-9158</contrib-id>
          <name>
            <surname>Cirenza</surname>
            <given-names>Claudio</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
        <contrib contrib-type="author">
          <name>
            <surname>Paola</surname>
            <given-names>Angelo Amato Vincenzo de</given-names>
          </name>
          <xref ref-type="aff" rid="aff1001">
            <sup>1</sup>
          </xref>
        </contrib>
      </contrib-group>
      <aff id="aff1001">
        <label>1</label>
        <country country="BR">Brazil</country>
        <institution content-type="original">Universidade Federal de S&#227;o Paulo Escola Paulista de Medicina, S&#227;o Paulo, SP - Brazil</institution>
      </aff>
      <author-notes>
        <corresp id="c01001">Mailing Address: Jos&#233; Nunes de Alencar Neto &#8226; Universidade Federal de S&#227;o Paulo Escola Paulista de Medicina - Rua Napole&#227;o de Barros, 715. Postal Code 04023-062, S&#227;o Paulo, SP &#8211; Brazil E-mail: josenunesalencar@gmail.com</corresp>
        <fn fn-type="con">
          <p>Author contributions</p>
          <p>Conception and design of the research: Alencar Neto JN, Cirenza C, Paola AAV; Acquisition of data: Alencar Neto JN, Sakai MH, Moraes SRR; Writing of the manuscript: Alencar Neto JN, Sakai MH, Moraes SRR, Paola AAV; Critical revision of the manuscript for intellectual content: Cirenza C, Paola AAV.</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>Heart Block</kwd>
        <kwd>Chest Pain</kwd>
        <kwd>Coronary Artery Diseases/physiopathology</kwd>
        <kwd>Cardiac Electrophysiology</kwd>
        <kwd>Electrocardiography</kwd>
        <kwd>Ecocardiography</kwd>
      </kwd-group>
    </front-stub>
    <body>
      <sec sec-type="intro">
        <title>Introduction</title>
        <p>The development of chest pain associated with intermittent left bundle branch block (LBBB) in the absence of coronary artery disease has been described in the literature as painful left bundle branch block syndrome. The mechanism responsible for the chest pain is unknown, but the main current hypothesis is related to acute cardiac dyssynchrony.</p>
        <p>In this syndrome, the LBBB occurs when the cycle length is equal to or less than the refractory period of the left bundle, mainly during physical effort. The chest pain in the case of the painful LBBB syndrome may range from a mild discomfort to a disabling condition.</p>
        <p>This report describes the case of a patient with typical rate-dependent LBBB associated with chest pain who was referred to electrophysiologic study (EPS) without evidence of arrhythmias.</p>
      </sec>
      <sec>
        <title>Case Report</title>
        <p>A 41-year-old woman with a history of controlled hypertension and a 2-year history of palpitations associated with chest pain triggered by minimal efforts during daily activity, which lasted up to 2 hours. The chest pain was described as a pressure sensation that radiated to the left arm associated with nausea and dyspnea. The episodes were characterized by sudden onset, without any prodromes, with spontaneous improvement. She was initially treated with atenolol 25 mg bid, with partial relief of symptoms. There was no family history of unexplained syncope or sudden cardiac death. Her physical examination was unremarkable. The 12-lead electrocardiogram (ECG) during crisis revealed a wide complex tachycardia with complete left bundle branch block (LBBB), with inferior axis and a P wave compatible with sinus rhythm. Even so, the patient was referred to EPS, which did not evidence arrhythmogenic substrates.</p>
        <p>However, at the start of a 600 miliseconds continuous atrial pacing, a rate-dependent LBBB was noted. Immediately succeeding the blockade of the LBBB, the patient, who was not maintained under sedation, started to complain about the same symptoms already described. The LBBB persisted during some minutes and relieved itself, concomitant to the relief of pain. The ECG is shown in <xref ref-type="fig" rid="f01001">Figure 1</xref> . It is a typical third degree LBBB with a 138 ms QRS complex duration, superior axis and a sinus P wave.</p>
        <p>
          <fig id="f01001">
            <label>Figure 1</label>
            <caption>
              <title>
                <italic><bold>&#8211;</bold> 12-lead ECG of the onset of pain immediately after left bundle branch block</italic>
              </title>
            </caption>
            <graphic xlink:href="1678-4170-abc-114-4suppl1-0034-90295-gf01-en.jpg"/></fig>
        </p>
        <p>The basal 12-lead ECG was normal ( <xref ref-type="fig" rid="f02001">Figure 2</xref> ). The 24-hour ECG Holter monitoring revealed that basal HR was between 56 and 116 bpm during daily activities, with no evidence of LBBB. Transthoracic echocardiography and cardiac magnetic resonance both showed normal systolic function with no myocardial or valve pathologies. All cardiac chambers were normal in size. A stress test evidenced the development of left bundle branch block associated with thoracic pain. The CT angiography discarded coronary artery disease and myocardial perfusion defects on dypiridamole. Currently, the patient is receiving atenolol 50 mg bid and no recurrence of palpitations or chest pain was evidenced at the 6-month follow-up.</p>
        <p>
          <fig id="f02001">
            <label>Figure 2</label>
            <caption>
              <title>
                <italic><bold>&#8211;</bold> Normal basal 12-lead ECG.</italic>
              </title>
            </caption>
            <graphic xlink:href="1678-4170-abc-114-4suppl1-0034-90295-gf02-en.jpg"/></fig>
        </p>
      </sec>
      <sec sec-type="discussion">
        <title>Discussion</title>
        <p>In 1946, the first report on intermittent left bundle branch block induced by effort was published. The patient presented with palpitations and aching feeling in precordium during crises. However, the coronary angiography was not performed due to the technology available at the time.<sup><xref ref-type="bibr" rid="B1">1</xref></sup> In 1976, Vieweg et al.<sup><xref ref-type="bibr" rid="B2">2</xref></sup> reported the first case of left bundle branch block associated with angina of effort, with angiographic evidence of normal coronary arteries. Although the patient was considered to have angina, atypical characteristics were present: sudden onset and offset, concomitantly with LBBB and after its disappearance, respectively.<sup><xref ref-type="bibr" rid="B2">2</xref></sup> In 1982, Virtanen et al. carried out a study with 7 patients with new left bundle branch block and chest pain during exercise test, all of them with normal coronary angiography. In this study, the patient&#8217;s pain pattern was evaluated and considered as atypical pain, because of sudden onset and offset.<sup><xref ref-type="bibr" rid="B3">3</xref></sup> Subsequently, new cases were reported, and this condition was called painful left bundle branch block syndrome.</p>
        <p>The mechanisms of painful LBBB syndrome are unclear. The possibility of demand ischemia due to coronary lesions or spasms was initially considered as a possible cause for this syndrome. However, this assumption was soon proved wrong. Immediate onset/offset of pain is incompatible with ischemia.<sup><xref ref-type="bibr" rid="B4">4</xref></sup> Nitroglycerin was proved to be ineffective<sup><xref ref-type="bibr" rid="B2">2</xref></sup> and sometimes induced LBBB due to tachycardia. Nuclear imaging is frequently negative in these patients and vasospasm has also been discarded.<sup><xref ref-type="bibr" rid="B5">5</xref> , <xref ref-type="bibr" rid="B6">6</xref></sup></p>
        <p>The best theory so far was proposed by Virtanen et al.,<sup><xref ref-type="bibr" rid="B3">3</xref></sup> who speculated that the pain could be induced by abnormal systolic motion of the septum in ventriculography. An inferior axis which presented uniformly in a series of cases made the authors hypothesize that this could reflect a specific contractility pattern. Shvlikin et al.<sup><xref ref-type="bibr" rid="B7">7</xref></sup> proposed criteria to the diagnosis of painful LBBB syndrome ( <xref ref-type="table" rid="t1001">Table 1</xref> ).<sup><xref ref-type="bibr" rid="B7">7</xref></sup></p>
        <p>
          <table-wrap id="t1001">
            <label>Table 1</label>
            <caption>
              <title>&#8211; Painful LBBB syndrome criteria</title>
            </caption>
            <table frame="hsides" rules="groups">
              <colgroup>
                <col/>
              </colgroup>
              <tbody>
                <tr>
                  <td align="center">Abrupt onset of chest pain with development of LBBB</td>
                </tr>
                <tr>
                  <td align="center">Simultaneous resolution of symptoms with resolution of LBBB (occasionally absent)</td>
                </tr>
                <tr>
                  <td align="center">Normal 12-lead ECG before and after LBBB</td>
                </tr>
                <tr>
                  <td align="center">Absence of myocardial ischemia during functional stress test</td>
                </tr>
                <tr>
                  <td align="center">Normal left ventricular function and absence of other conditions that may explain the symptoms</td>
                </tr>
                <tr>
                  <td align="center">Low precordial S/T wave ratio (&lt; 1.8) and inferior axis</td>
                </tr>
              </tbody>
            </table>
            <table-wrap-foot>
              <fn id="TFN1001">
                <p>
                  <italic>Criteria proposed for the diagnosis of the painful LBBB syndrome. Adapted from Shvilkin.<sup>7</sup></italic>
                </p>
              </fn>
            </table-wrap-foot>
          </table-wrap>
        </p>
        <p>Similar to the memory T waves of paced patients, chronic LBBB has lower amplitude T waves than acute LBBB. In a prospective study, a S/T ratio &lt; 2.5 in precordial leads proved to be useful (100% sensitivity and 89% specificity) to help distinguish between new-onset or chronic LBBB,<sup><xref ref-type="bibr" rid="B8">8</xref></sup> which refers to one of the items of the criteria proposed in <xref ref-type="table" rid="t1001">Table 1</xref> .</p>
        <p>The patient described in this paper was referred to EPS because of the mistaken hypothesis of supraventricular tachycardia with aberrancy. During the study, with continuous atrial pacing, we had the opportunity to register the exact moment of left bundle branch blockade and the immediate complain about the same pain she referred to as chronic.</p>
        <p>Regarding the criteria proposed by Schvilkin et al.,<sup><xref ref-type="bibr" rid="B8">8</xref></sup> our case matches all but one criterion: the &#8220;inferior axis criterion&#8221;. However, other publications evidenced superior QRS complex axis as well.<sup><xref ref-type="bibr" rid="B9">9</xref> , <xref ref-type="bibr" rid="B10">10</xref></sup> The S/T wave ratio was 1,33 in V2 ( <xref ref-type="fig" rid="f03001">Figure 3</xref> ), which is compatible with acute onset LBBB. The patient had abrupt onset of pain, as registered by the members of our team in our electrophysiology lab; the resolution of the symptoms occurred immediately after the resolution of the LBBB; The basal 12-lead ECG was normal. A stress test discarded myocardial ischemia and the CT angiography evidenced normal coronary arteries. Echocardiography and cardiac resonance were both normal, excluding secondary causes of angina.</p>
        <p>
          <fig id="f03001">
            <label>Figure 3</label>
            <caption>
              <title>
                <italic><bold>&#8211;</bold> S/T ratio &lt; 1.8 in V2.</italic>
              </title>
            </caption>
            <graphic xlink:href="1678-4170-abc-114-4suppl1-0034-90295-gf03-en.jpg"/></fig>
        </p>
      </sec>
      <sec sec-type="conclusions">
        <title>Conclusion</title>
        <p>We reported the case of a patient with painful LBBB who was referred to electrophysiology study. The abrupt onset of pain immediate after left bundle blockade is incompatible with ischemia and the patient underwent exams that discarded coronary and myocardial involvement. The best hypothesis for the pathophysiology of this syndrome is painful desynchrony of the heart due to acute onset LBBB. To our knowledge, this is the first case report of this syndrome in a Brazilian medical journal.</p>
      </sec>
    </body>
    <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 fn-type="other">
          <p>Ethics approval and consent to participate</p>
          <p>This article does not contain any studies with human participants or animals performed by any of the authors.</p>
        </fn>
      </fn-group>
      <fn-group>
        <fn fn-type="supported-by">
          <p>Sources of Funding</p>
          <p>There were no external funding sources for this study.</p>
        </fn>
      </fn-group>
    </back>
  </sub-article>
</article>
