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    <front>
        <journal-meta>
            <journal-id journal-id-type="pmc">F1000Research</journal-id>
            <journal-title-group>
                <journal-title>F1000Research</journal-title>
            </journal-title-group>
            <issn pub-type="epub">2046-1402</issn>
            <publisher>
                <publisher-name>F1000 Research Limited</publisher-name>
                <publisher-loc>London, UK</publisher-loc>
            </publisher>
        </journal-meta>
        <article-meta>
            <article-id pub-id-type="doi">10.12688/f1000research.148440.1</article-id>
            <article-categories>
                <subj-group subj-group-type="heading">
                    <subject>Case Report</subject>
                </subj-group>
                <subj-group>
                    <subject>Articles</subject>
                </subj-group>
            </article-categories>
            <title-group>
                <article-title>Case Report: Percutaneous intercostal artery catheterization to treat a difficult-to-access spinal dural arteriovenous fistula</article-title>
                <fn-group content-type="pub-status">
                    <fn>
                        <p>[version 1; peer review: awaiting peer review]</p>
                    </fn>
                </fn-group>
            </title-group>
            <contrib-group>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Martiel</surname>
                        <given-names>Manrique-Zegarra</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Software</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <uri content-type="orcid">https://orcid.org/0000-0002-1556-9975</uri>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Carlos</surname>
                        <given-names>Toledano-Ill&#x00e1;n</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Maria Jesus</surname>
                        <given-names>Garcia-Sanchez</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Jorge</surname>
                        <given-names>Escart&#x00ed;n L&#x00f3;pez</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Josep</surname>
                        <given-names>Puig</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a2">2</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Alex</surname>
                        <given-names>L&#x00fc;ttich</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <xref ref-type="aff" rid="a3">3</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Ignacio</surname>
                        <given-names>Muguruza Trueba</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a4">4</xref>
                </contrib>
                <contrib contrib-type="author" corresp="yes">
                    <name>
                        <surname>Miguel A.</surname>
                        <given-names>Vences</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="corresp" rid="c1">a</xref>
                    <xref ref-type="aff" rid="a1">1</xref>
                    <xref ref-type="aff" rid="a5">5</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Claudio</surname>
                        <given-names>Rodriguez-Fernandez</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <aff id="a1">
                    <label>1</label>Jimenez Diaz Foundation University Hospital Interventional Neuroradiology Service, Madrid, Community of Madrid, Spain</aff>
                <aff id="a2">
                    <label>2</label>Department of Radiology, Hospital Universitari Dr Josep Trueta, Girona, Spain</aff>
                <aff id="a3">
                    <label>3</label>Department of Interventional Neuroradiology, Hospital Universitario de Donostia, San Sebasti&#x00e1;n, Basque Country, Spain</aff>
                <aff id="a4">
                    <label>4</label>department of Thoracic Surgery, Hospital Universitario Rey Juan Carlos, Madrid, Spain</aff>
                <aff id="a5">
                    <label>5</label>Medicine School, Cesar Vallejo University, Piura, Peru</aff>
            </contrib-group>
            <author-notes>
                <corresp id="c1">
                    <label>a</label>
                    <email xlink:href="mailto:mavences@ucvvirtual.edu.pe">mavences@ucvvirtual.edu.pe</email>
                </corresp>
                <fn fn-type="conflict">
                    <p>No competing interests were disclosed.</p>
                </fn>
            </author-notes>
            <pub-date pub-type="epub">
                <day>30</day>
                <month>4</month>
                <year>2024</year>
            </pub-date>
            <pub-date pub-type="collection">
                <year>2024</year>
            </pub-date>
            <volume>13</volume>
            <elocation-id>430</elocation-id>
            <history>
                <date date-type="accepted">
                    <day>25</day>
                    <month>4</month>
                    <year>2024</year>
                </date>
            </history>
            <permissions>
                <copyright-statement>Copyright: &#x00a9; 2024 Martiel MZ et al.</copyright-statement>
                <copyright-year>2024</copyright-year>
                <license xlink:href="https://creativecommons.org/licenses/by/4.0/">
                    <license-p>This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
                </license>
            </permissions>
            <self-uri content-type="pdf" xlink:href="https://f1000research.com/articles/13-430/pdf"/>
            <abstract>
                <p>Spinal dural arteriovenous fistulas (SDAVF) are a subtype of spinal arteriovenous malformation (AVM). Although rare, SDAVF are the most common spinal vascular malformations and have potential devastating neurologic consequences, sometimes irreversible. Originally, they were treated with surgery alone, however endovascular treatment of SDAVF has evolved recently with good results and the understanding of the anatomy plays a key role. The feeder vessel of the AV shunt is supplied by the radiculomedullary or radiculopial artery, the shunt is usually located within the root sleeve of the dura and the SDAVF is drained by the medullary vein via retrograde flow. Generally the endovascular approach uses the catheterization of the segmental arteries; however, aortic thoracolumbar aneurysms can render endovascular access difficult and risky. We present a case where an SDAVF in woman with aneurysmal dilation of the descending aorta and a mural thrombus was successfully treated through percutaneous catheterization of intercostal artery.</p>
            </abstract>
            <kwd-group kwd-group-type="author">
                <kwd>Spine</kwd>
                <kwd>arteriovenous fistula</kwd>
                <kwd>Peripheral Catheterization (source: MeSH).</kwd>
            </kwd-group>
            <funding-group>
                <funding-statement>The author(s) declared that no grants were involved in supporting this work.</funding-statement>
            </funding-group>
        </article-meta>
    </front>
    <body>
        <sec id="sec1" sec-type="intro">
            <title>Introduction</title>
            <p>Spinal dural arteriovenous fistulas (SDAVF) account for 70% of spinal vascular malformations. In SDAVFs, a direct shunt between a radiculomeningeal artery and a radicular vein results in increased venous pressure and decreased drainage of normal spinal veins, leading to venous congestion, intramedullary edema, chronic hypoxia, and myelopathy. In 90% of SDAVFs, the shunt is located in the lateral epidural space where the radicular vein passes through the dura at the dorsal surface of the dura root sleeve in the intervertebral foramen.
                <sup>
                    <xref ref-type="bibr" rid="ref1">1</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref2">2</xref>
                </sup>
            </p>
        </sec>
        <sec id="sec2">
            <title>Case report</title>
            <sec id="sec3">
                <title>Patient information</title>
                <p>An adult patient with no relevant medical history presented with intermittent upper and lower limb paresthesias and motor deficits, sometimes associated with cold sweats, nausea, and palpitations. She had no fever, recent trauma, or incontinence.</p>
                <p>Physical examination revealed right lower-limb weakness and hyperalgesia.</p>
            </sec>
            <sec id="sec4">
                <title>Diagnostic assessment</title>
                <p>CTA showed aneurysmal dilation of the descending aorta with a mural thrombus. T2-weighted MRI sequences showed centromedullary edema over multiple spinal cord segments and dilated and coiled posterolateral perimedullary vessels. MRA done to locate the suspected SDAVF showed early venous filling with an arteriovenous shunt at the right T10 level (
                    <xref ref-type="fig" rid="f1">Figure 1</xref>).</p>
                <fig fig-type="figure" id="f1" orientation="portrait" position="float">
                    <label>Figure 1. </label>
                    <caption>
                        <title>Imaging protocol.</title>
                        <p>[A] Volume rendering reconstruction from CTA shows the aneurysm and associated mural thrombus in the descending aorta; Sagittal [B] and axial [C] T2-weighted MRI show centromedullary edema and dilated perimedullary vessels. Axial [D] and coronal [E] first-pass contrast-enhanced MRA images show early venous filling (arrowheads) and the presumed shunt point (arrow) at the right T10 level.</p>
                    </caption>
                    <graphic id="gr1" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/162749/42018aa3-a0e4-45e7-b6f3-91a392167c30_figure1.gif"/>
                </fig>
                <p>DSA showed delayed venous return in the great anterior radiculomedullary artery originating from the right T11 segmental artery (not shown); however, the partially thrombosed aneurysmatic dilation of the aorta and the morphology and disposition of the thoracic segment precluded the catheterization of some lower thoracic segmental arteries.</p>
            </sec>
            <sec id="sec5">
                <title>Therapeutic intervention</title>
                <p>Surgical repair failed because the intradural vein receiving blood from the shunt could not be identified. The interdisciplinary team decided on a combined surgical and endovascular approach in which a minimal partial posterolateral rib resection with the patient in left lateral decubitus position exposed the ventral branch of the 10th right posterior intercostal artery (
                    <xref ref-type="fig" rid="f2">Figure 2</xref>), which was catheterized with a 4F introducer set (4F Micro-stick introducer set, Medcomp) and a 0.014 inch microwire. DSA revealed early venous filling and retrograde contrast uptake of the upper and lower radiculomedullary veins, confirming the diagnosis of SDAVF (
                    <xref ref-type="fig" rid="f3">Figure 3</xref>).</p>
                <fig fig-type="figure" id="f2" orientation="portrait" position="float">
                    <label>Figure 2. </label>
                    <caption>
                        <title>Therapeutical procedure.</title>
                        <p>[A] Minimal partial posterolateral rib resection with the patient in left lateral decubitus to expose the ventral branch of the 10th right posterior intercostal artery. [B, C] Catheterization of the ventral branch of the 10th right posterior intercostal artery with a micropuncture set and a 0.014-inch microwire.</p>
                    </caption>
                    <graphic id="gr2" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/162749/42018aa3-a0e4-45e7-b6f3-91a392167c30_figure2.gif"/>
                </fig>
                <fig fig-type="figure" id="f3" orientation="portrait" position="float">
                    <label>Figure 3. </label>
                    <caption>
                        <title>Endovascular procedure.</title>
                        <p>[A] Unsubstracted fluoroscopic image showing the intercostal access and opacification of the Spinal dural arteriovenous fistulas (SDAVF) at the T 10 level. Superselective injection and 3D rotational DSA with MIP reconstruction [B] show the feeding artery (arrowhead), the shunting zone (arrow), and the proximal draining vein (asterisk). [C, D] Exclusion of the SDAVF after embolization and coil placement in the segmentary artery. [E] Sagittal Short tau inversion recovery (STIR) MRI image 3-months after treatment showing less evident cord edema.</p>
                    </caption>
                    <graphic id="gr3" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/162749/42018aa3-a0e4-45e7-b6f3-91a392167c30_figure3.gif"/>
                </fig>
                <p>Injection from the segmental artery verified the absence of supply to the spinal cord from the pedicle feeding the SDAVF. After 3D-rotational DSA to better characterize the lesion, we injected approximately 0.5 cc of ethylene vinyl alcohol copolymer (SQUID-18, Emboflu; Gland, Switzerland, supplied by Balt) into the fistula with little pass to the draining vein. Finally, we placed a coil in the segmentary artery to mark the level of the fistula. Control images demonstrated the exclusion of the fistula and patency of the segmental artery (
                    <xref ref-type="fig" rid="f3">Figure 3</xref>).</p>
            </sec>
            <sec id="sec6">
                <title>Follow-up and outcomes</title>
                <p>The patient was discharged without complications after 2 days.</p>
                <p>At follow-up, her lower limb strength had improved and on follow-up MRI three months after procedure (
                    <xref ref-type="fig" rid="f3">Figure 3</xref>) the prominent perimedullary vessels and cord edema were less evident. Even though there was not complete pass to the draining vein, it was enough to diminish the flow to the fistula and to get symptomatic relief.</p>
            </sec>
        </sec>
        <sec id="sec7" sec-type="discussion">
            <title>Discussion</title>
            <p>Treatment for SDAVF aims to occlude the shunt. Surgical exclusion of the fistula by a classic approach or minimally invasive techniques yields excellent results,
                <sup>
                    <xref ref-type="bibr" rid="ref3">3</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref4">4</xref>
                </sup> better than the endovascular approach.</p>
            <p>Different results have been reported with endovascular treatment with different embolic materials.
                <sup>
                    <xref ref-type="bibr" rid="ref5">5</xref>
                </sup>
                <sup>&#x2013;</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref7">7</xref>
                </sup> In our institution, endovascular treatment is considered for spinal arteriovenous shunts whenever safely possible. After superselective catheterization of the feeding artery, a liquid embolic agent is injected through the fistula to occlude the proximal segment of the draining vein to prevent subsequent intradural collateral filling of the fistula.
                <sup>
                    <xref ref-type="bibr" rid="ref6">6</xref>
                </sup>
            </p>
            <p>In our case, MRI suggested SDAVF and MRA suggested the location, but the diagnosis could not be confirmed with conventional spinal DSA because the partially thrombosed thoracic aortic aneurysm and the morphology of the lower thoracic aorta made it impossible to catheterize the segmental arteries at the level of the suspected shunt. After surgical repair failed, a different approach was necessary.</p>
            <p>The ventral branch of the posterior intercostal artery runs circumferentially under the rib; the dorsal branch has a spinal branch that enters the vertebral canal through the intervertebral foramen and gives rise to the radicular artery, among others.
                <sup>
                    <xref ref-type="bibr" rid="ref8">8</xref>
                </sup>
            </p>
            <p>We used a novel technique to access the ventral branch of the posterior intercostal artery to catheterize the dorsal branch, confirm the SDAVF, and embolize the lesion after superselective catheterization.</p>
            <sec id="sec8">
                <title>Patient perspective</title>
                <p>&#x201c;My family and I had a bad time when the neurologic symptoms started and progressed. I had great support during and after the treatment. I&#x2019;m walking again and taking care of my grandchildren. I&#x2019;m really grateful with Dr. Rodr&#x00ed;guez and his team.&#x201d;</p>
            </sec>
        </sec>
        <sec id="sec9">
            <title>Authors&#x2019; contributions</title>
            <p>All authors participated in design, writing, critical review and approved the final version of the manuscript.</p>
        </sec>
        <sec id="sec10">
            <title>Ethics statement</title>
            <p>The present study is a case report, so it does not need ethical approval. The authors received signed informed consent from the patient and are committed to respecting bioethical research principles as well as the Declaration of Helsinki.</p>
        </sec>
        <sec id="sec11">
            <title>Consent to publish</title>
            <p>We obtained the patient&#x2019;s written informed consent to participate in this study. Also, the patient gave us written permission for the publication of images and data included in this case report.</p>
        </sec>
    </body>
    <back>
        <sec id="sec14" sec-type="data-availability">
            <title>Data availability statement</title>
            <p>All data underlying the results are available as part of the article and no additional source data are required.</p>
        </sec>
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