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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Paget-Schroetter syndrome in the absence of common predisposing factors: a case report</title><author><name sortKey="Ibrahim, Ramy" sort="Ibrahim, Ramy" uniqKey="Ibrahim R" first="Ramy" last="Ibrahim">Ramy Ibrahim</name><affiliation><nlm:aff id="Aff1">Premier Medical Associates Group Orlando, Florida, USA</nlm:aff></affiliation></author><author><name sortKey="Dashkova, Irina" sort="Dashkova, Irina" uniqKey="Dashkova I" first="Irina" last="Dashkova">Irina Dashkova</name><affiliation><nlm:aff id="Aff2"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Division of Geriatric and Palliative Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</nlm:aff></affiliation></author><author><name sortKey="Williams, Myia" sort="Williams, Myia" uniqKey="Williams M" first="Myia" last="Williams">Myia Williams</name><affiliation><nlm:aff id="Aff3"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</nlm:aff></affiliation></author><author><name sortKey="Kozikowski, Andrzej" sort="Kozikowski, Andrzej" uniqKey="Kozikowski A" first="Andrzej" last="Kozikowski">Andrzej Kozikowski</name><affiliation><nlm:aff id="Aff4"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>NewYork, USA</nlm:aff></affiliation></author><author><name sortKey="Abrol, Neeraj" sort="Abrol, Neeraj" uniqKey="Abrol N" first="Neeraj" last="Abrol">Neeraj Abrol</name><affiliation><nlm:aff id="Aff5">Attending Physician, Brookdale Hospital, New York, USA</nlm:aff></affiliation></author><author><name sortKey="Gandhi, Anjula" sort="Gandhi, Anjula" uniqKey="Gandhi A" first="Anjula" last="Gandhi">Anjula Gandhi</name><affiliation><nlm:aff id="Aff6">Internal Medicine Residency Program, Brookdale Hospital, New York, USA</nlm:aff></affiliation></author><author><name sortKey="Pekmezaris, Renee" sort="Pekmezaris, Renee" uniqKey="Pekmezaris R" first="Renee" last="Pekmezaris">Renee Pekmezaris</name><affiliation><nlm:aff id="Aff7"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">28781584</idno><idno type="pmc">5537934</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5537934</idno><idno type="RBID">PMC:5537934</idno><idno type="doi">10.1186/s12959-017-0146-0</idno><date when="2017">2017</date><idno type="wicri:Area/Pmc/Corpus">000A54</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000A54</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Paget-Schroetter syndrome in the absence of common predisposing factors: a case report</title><author><name sortKey="Ibrahim, Ramy" sort="Ibrahim, Ramy" uniqKey="Ibrahim R" first="Ramy" last="Ibrahim">Ramy Ibrahim</name><affiliation><nlm:aff id="Aff1">Premier Medical Associates Group Orlando, Florida, USA</nlm:aff></affiliation></author><author><name sortKey="Dashkova, Irina" sort="Dashkova, Irina" uniqKey="Dashkova I" first="Irina" last="Dashkova">Irina Dashkova</name><affiliation><nlm:aff id="Aff2"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Division of Geriatric and Palliative Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</nlm:aff></affiliation></author><author><name sortKey="Williams, Myia" sort="Williams, Myia" uniqKey="Williams M" first="Myia" last="Williams">Myia Williams</name><affiliation><nlm:aff id="Aff3"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</nlm:aff></affiliation></author><author><name sortKey="Kozikowski, Andrzej" sort="Kozikowski, Andrzej" uniqKey="Kozikowski A" first="Andrzej" last="Kozikowski">Andrzej Kozikowski</name><affiliation><nlm:aff id="Aff4"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>NewYork, USA</nlm:aff></affiliation></author><author><name sortKey="Abrol, Neeraj" sort="Abrol, Neeraj" uniqKey="Abrol N" first="Neeraj" last="Abrol">Neeraj Abrol</name><affiliation><nlm:aff id="Aff5">Attending Physician, Brookdale Hospital, New York, USA</nlm:aff></affiliation></author><author><name sortKey="Gandhi, Anjula" sort="Gandhi, Anjula" uniqKey="Gandhi A" first="Anjula" last="Gandhi">Anjula Gandhi</name><affiliation><nlm:aff id="Aff6">Internal Medicine Residency Program, Brookdale Hospital, New York, USA</nlm:aff></affiliation></author><author><name sortKey="Pekmezaris, Renee" sort="Pekmezaris, Renee" uniqKey="Pekmezaris R" first="Renee" last="Pekmezaris">Renee Pekmezaris</name><affiliation><nlm:aff id="Aff7"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</nlm:aff></affiliation></author></analytic><series><title level="j">Thrombosis Journal</title><idno type="eISSN">1477-9560</idno><imprint><date when="2017">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec><title>Background</title><p id="Par1">Paget-Schrotter Syndrome (PSS) also known as “effort thrombosis” is a form of primary thrombosis in the subclavian vein at the costoclavicular junction is usually seen in younger patients after repeated strenuous activity of the shoulders and arms. When occurring in younger patients, PSS presents itself with predisposing factors such as unilateral dull, aching pain in the shoulder or axilla and swelling of the arm and hand.</p></sec><sec><title>Case Presentation</title><p id="Par2">We report a rare case of unusual left axillo-subclavian vein thrombosis in absence of clear risk factors and a negative hypercoagulable workup in a 36-year-old Hispanic woman who presented with 2 days duration of left upper extremity pain and swelling after a week of heavy exercise in aerobic class. Very few documented cases of primary or spontaneous ASVT in absence of clear factors and in such anatomical location have been previously reported.</p><p id="Par3">The patient was started on strict precautions of left upper extremity immobilization, analgesics in the form of Tylenol 650 mg every 6 h for pain as well as cold compresses. Lovenox 90 mg subcutaneous twice daily (1 mg/kg BID) was started together with warfarin to keep INR 2–3.</p></sec><sec><title>Conclusion</title><p id="Par4">In addition to the unusual location in the left upper extremity in our case, the absence of common etiologic factors makes our case of Paget-Schroetter Syndrome a very unique one. Presently, there is a lack of guided management of rare conditions such as our case, or consensus among the sources. Physicians should be aware of this rare disease since untreated conditions may be debilitating for the patient and very costly especially if diagnosed with a delay.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct></biblStruct><biblStruct><analytic><author><name sortKey="Alla, Vm" uniqKey="Alla V">VM Alla</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Von Schroetter, L" uniqKey="Von Schroetter L">L von Schroetter</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Martinelli, I" uniqKey="Martinelli I">I Martinelli</name></author><author><name sortKey="Mannucci, Pm" uniqKey="Mannucci P">PM Mannucci</name></author><author><name sortKey="De Stefano, V" uniqKey="De Stefano V">V De Stefano</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mustafa, S" uniqKey="Mustafa S">S Mustafa</name></author><author><name sortKey="Stein, Pd" uniqKey="Stein P">PD Stein</name></author><author><name sortKey="Patel, Kc" uniqKey="Patel K">KC Patel</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Urschel, Hc" uniqKey="Urschel H">HC Urschel</name></author><author><name sortKey="Patel, An" uniqKey="Patel A">AN Patel</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kommareddy, A" uniqKey="Kommareddy A">A Kommareddy</name></author><author><name sortKey="Zaroukian, Mh" uniqKey="Zaroukian M">MH Zaroukian</name></author><author><name sortKey="Hassouna, Hi" uniqKey="Hassouna H">HI Hassouna</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Joffe, Hv" uniqKey="Joffe H">HV Joffe</name></author><author><name sortKey="Kucher, N" uniqKey="Kucher N">N Kucher</name></author><author><name sortKey="Tapson, Vf" uniqKey="Tapson V">VF Tapson</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Louis, J" uniqKey="Louis J">J Louis</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Zell, L" uniqKey="Zell L">L Zell</name></author><author><name sortKey="Kindermann, W" uniqKey="Kindermann W">W Kindermann</name></author><author><name sortKey="Marschall, F" uniqKey="Marschall F">F Marschall</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kooij, Jd" uniqKey="Kooij J">JD Kooij</name></author><author><name sortKey="Van Der Zant, Fm" uniqKey="Van Der Zant F">FM van der Zant</name></author><author><name sortKey="Van Beek, Ej" uniqKey="Van Beek E">EJ van Beek</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Monreal, M" uniqKey="Monreal M">M Monreal</name></author><author><name sortKey="Lafoz, E" uniqKey="Lafoz E">E Lafoz</name></author><author><name sortKey="Ruiz, J" uniqKey="Ruiz J">J Ruiz</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Prandoni, P" uniqKey="Prandoni P">P Prandoni</name></author><author><name sortKey="Polistena, P" uniqKey="Polistena P">P Polistena</name></author><author><name sortKey="Bernardi, E" uniqKey="Bernardi E">E Bernardi</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Elman, Ee" uniqKey="Elman E">EE Elman</name></author><author><name sortKey="Kahn, Sr" uniqKey="Kahn S">SR Kahn</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Prandoni, P" uniqKey="Prandoni P">P Prandoni</name></author><author><name sortKey="Polistena, P" uniqKey="Polistena P">P Polistena</name></author><author><name sortKey="Bernardi, E" uniqKey="Bernardi E">E Bernardi</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hughes, Es" uniqKey="Hughes E">ES HUGHES</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Qaseem, A" uniqKey="Qaseem A">A Qaseem</name></author><author><name sortKey="Snow, V" uniqKey="Snow V">V Snow</name></author><author><name sortKey="Barry, P" uniqKey="Barry P">P Barry</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Thromb J</journal-id><journal-id journal-id-type="iso-abbrev">Thromb J</journal-id><journal-title-group><journal-title>Thrombosis Journal</journal-title></journal-title-group><issn pub-type="epub">1477-9560</issn><publisher><publisher-name>BioMed Central</publisher-name><publisher-loc>London</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">28781584</article-id><article-id pub-id-type="pmc">5537934</article-id><article-id pub-id-type="publisher-id">146</article-id><article-id pub-id-type="doi">10.1186/s12959-017-0146-0</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Paget-Schroetter syndrome in the absence of common predisposing factors: a case report</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Ibrahim</surname><given-names>Ramy</given-names></name><xref ref-type="aff" rid="Aff1">1</xref></contrib><contrib contrib-type="author"><name><surname>Dashkova</surname><given-names>Irina</given-names></name><xref ref-type="aff" rid="Aff2">2</xref></contrib><contrib contrib-type="author" corresp="yes"><contrib-id contrib-id-type="orcid">http://orcid.org/0000-0002-6295-8391</contrib-id><name><surname>Williams</surname><given-names>Myia</given-names></name><address><email>mwilliam26@northwell.edu</email></address><xref ref-type="aff" rid="Aff3">3</xref></contrib><contrib contrib-type="author"><name><surname>Kozikowski</surname><given-names>Andrzej</given-names></name><xref ref-type="aff" rid="Aff4">4</xref></contrib><contrib contrib-type="author"><name><surname>Abrol</surname><given-names>Neeraj</given-names></name><xref ref-type="aff" rid="Aff5">5</xref></contrib><contrib contrib-type="author"><name><surname>Gandhi</surname><given-names>Anjula</given-names></name><xref ref-type="aff" rid="Aff6">6</xref></contrib><contrib contrib-type="author"><name><surname>Pekmezaris</surname><given-names>Renee</given-names></name><xref ref-type="aff" rid="Aff7">7</xref></contrib><aff id="Aff1"><label>1</label>Premier Medical Associates Group Orlando, Florida, USA</aff><aff id="Aff2"><label>2</label><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Division of Geriatric and Palliative Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</aff><aff id="Aff3"><label>3</label><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</aff><aff id="Aff4"><label>4</label><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>NewYork, USA</aff><aff id="Aff5"><label>5</label>Attending Physician, Brookdale Hospital, New York, USA</aff><aff id="Aff6"><label>6</label>Internal Medicine Residency Program, Brookdale Hospital, New York, USA</aff><aff id="Aff7"><label>7</label><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2168 3646</institution-id><institution-id institution-id-type="GRID">grid.416477.7</institution-id><institution>Department of Medicine,</institution><institution>Northwell Health,</institution></institution-wrap>New York, USA</aff></contrib-group><pub-date pub-type="epub"><day>1</day><month>8</month><year>2017</year></pub-date><pub-date pub-type="pmc-release"><day>1</day><month>8</month><year>2017</year></pub-date><pub-date pub-type="collection"><year>2017</year></pub-date><volume>15</volume><elocation-id>20</elocation-id><history><date date-type="received"><day>3</day><month>4</month><year>2017</year></date><date date-type="accepted"><day>12</day><month>7</month><year>2017</year></date></history><permissions><copyright-statement>© The Author(s). 2017</copyright-statement><license license-type="OpenAccess"><license-p><bold>Open Access</bold>This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by/4.0/">http://creativecommons.org/licenses/by/4.0/</ext-link>), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/publicdomain/zero/1.0/">http://creativecommons.org/publicdomain/zero/1.0/</ext-link>) applies to the data made available in this article, unless otherwise stated.</license-p></license></permissions><abstract id="Abs1"><sec><title>Background</title><p id="Par1">Paget-Schrotter Syndrome (PSS) also known as “effort thrombosis” is a form of primary thrombosis in the subclavian vein at the costoclavicular junction is usually seen in younger patients after repeated strenuous activity of the shoulders and arms. When occurring in younger patients, PSS presents itself with predisposing factors such as unilateral dull, aching pain in the shoulder or axilla and swelling of the arm and hand.</p></sec><sec><title>Case Presentation</title><p id="Par2">We report a rare case of unusual left axillo-subclavian vein thrombosis in absence of clear risk factors and a negative hypercoagulable workup in a 36-year-old Hispanic woman who presented with 2 days duration of left upper extremity pain and swelling after a week of heavy exercise in aerobic class. Very few documented cases of primary or spontaneous ASVT in absence of clear factors and in such anatomical location have been previously reported.</p><p id="Par3">The patient was started on strict precautions of left upper extremity immobilization, analgesics in the form of Tylenol 650 mg every 6 h for pain as well as cold compresses. Lovenox 90 mg subcutaneous twice daily (1 mg/kg BID) was started together with warfarin to keep INR 2–3.</p></sec><sec><title>Conclusion</title><p id="Par4">In addition to the unusual location in the left upper extremity in our case, the absence of common etiologic factors makes our case of Paget-Schroetter Syndrome a very unique one. Presently, there is a lack of guided management of rare conditions such as our case, or consensus among the sources. Physicians should be aware of this rare disease since untreated conditions may be debilitating for the patient and very costly especially if diagnosed with a delay.</p></sec></abstract><kwd-group xml:lang="en"><title>Keywords</title><kwd>Deep Vein Thrombosis</kwd><kwd>Paget-Schrotter Syndrome</kwd><kwd>Axillary-subclavian venous thrombosis</kwd><kwd>Pulmonary Embolism</kwd><kwd>Thrombosis</kwd><kwd>Upper Extremity Deep Vein Thrombosis</kwd></kwd-group><custom-meta-group><custom-meta><meta-name>issue-copyright-statement</meta-name><meta-value>© The Author(s) 2017</meta-value></custom-meta></custom-meta-group></article-meta></front><body><sec id="Sec1"><title>Background</title><p id="Par15">Paget- Schroetter Syndrome involves axillary-subclavian venous thrombosis (ASVT) associated with strenuous and repeated activity of the upper extremities [<xref ref-type="bibr" rid="CR1">1</xref>, <xref ref-type="bibr" rid="CR2">2</xref>]. PSS is also called “effort” thrombosis denoting that the syndrome often occurs in physically active individuals after unusual strenuous use of the arm and shoulder [<xref ref-type="bibr" rid="CR3">3</xref>]. In addition, PSS is also referred to as “spontaneous” ASVT, highlighting its often dramatic, unexpected presentation in otherwise healthy young individuals [<xref ref-type="bibr" rid="CR4">4</xref>, <xref ref-type="bibr" rid="CR5">5</xref>]. On average, PSS accounts for at least 10–20% of upper extremity deep thrombosis and at least up to 30–40% of spontaneous axillary-subclavian venous thrombosis [<xref ref-type="bibr" rid="CR1">1</xref>, <xref ref-type="bibr" rid="CR2">2</xref>].</p><p id="Par16">PSS is usually experienced after sporting activities such as swimming, wrestling and gymnastics which often involve vigorous and continued movements of the upper extremity [<xref ref-type="bibr" rid="CR2">2</xref>]. It is believed that retroversion, hyperabduction and extension of the arm involved in strenuous sporting activities impose undue strain on the subclavian vein; which leads to micro-trauma of the endothelium and activation of the coagulation cascade. Consequently, it is not unusual that PSS usually occurs in the dominant arm of young, healthy and active men [<xref ref-type="bibr" rid="CR2">2</xref>]. Patients with PSS are customarily symptomatic, with swelling and arm discomfort being the most frequently reported and presented problems, as shown in Table <xref rid="Tab1" ref-type="table">1</xref> [<xref ref-type="bibr" rid="CR2">2</xref>, <xref ref-type="bibr" rid="CR6">6</xref>–<xref ref-type="bibr" rid="CR8">8</xref>]. Other symptoms reported include heaviness, redness of arm, cyanosis and dilated, visible veins across the shoulder and upper arm (Urschel’s sign) [<xref ref-type="bibr" rid="CR2">2</xref>, <xref ref-type="bibr" rid="CR6">6</xref>]. Often the symptom onset is either acute or sub-acute, however, occasionally, patients can present with chronic symptoms [<xref ref-type="bibr" rid="CR2">2</xref>]. In addition, not uncommon symptoms can present as nonspecific and at times mimic those of a muscular strain [<xref ref-type="bibr" rid="CR2">2</xref>, <xref ref-type="bibr" rid="CR9">9</xref>]. Further, a majority of patients have reported a discrete precipitating event of a sports related arm exertion. Similarly, trivial and harmless daily activities can result in PSS [<xref ref-type="bibr" rid="CR2">2</xref>, <xref ref-type="bibr" rid="CR6">6</xref>, <xref ref-type="bibr" rid="CR10">10</xref>].<table-wrap id="Tab1"><label>Table 1</label><caption><p>Manifestations of DVT</p></caption><table frame="hsides" rules="groups"><thead><tr><th>Type of Manifestation</th><th></th></tr></thead><tbody><tr><td>Asymptomatic</td><td></td></tr><tr><td>Pain in the arm, neck and shoulder region Diffuse arm swelling</td><td></td></tr><tr><td>Discoloration, tenderness and distension on the affected limb</td><td></td></tr><tr><td>Visible collaterals on the affected arm Arm discoloration and palpable vessels</td><td></td></tr></tbody></table></table-wrap></p><p id="Par17">Complications also seen in patients with PSS include pulmonary embolism (PE), post thrombotic syndrome and recurrent thrombosis [<xref ref-type="bibr" rid="CR2">2</xref>, <xref ref-type="bibr" rid="CR6">6</xref>, <xref ref-type="bibr" rid="CR7">7</xref>]. It is important to note there are mixed reports of lower incidence of PE in upper extremity DVT when compared with lower extremity DVT and catheter related UEDVT [<xref ref-type="bibr" rid="CR2">2</xref>, <xref ref-type="bibr" rid="CR8">8</xref>, <xref ref-type="bibr" rid="CR11">11</xref>, <xref ref-type="bibr" rid="CR12">12</xref>]. Regardless, health practitioners should bear in mind that the risk of PE in patients with PSS is real and significant [<xref ref-type="bibr" rid="CR2">2</xref>, <xref ref-type="bibr" rid="CR6">6</xref>, <xref ref-type="bibr" rid="CR7">7</xref>, <xref ref-type="bibr" rid="CR13">13</xref>, <xref ref-type="bibr" rid="CR14">14</xref>].</p><p id="Par18">We report a rare case of unusual left axillosubclavian vein thrombosis in absence of clear risk factors and a negative hypercoagulable workup in a 36-year-old Hispanic woman who presented with 2 days duration of left upper extremity pain and swelling after a week of heavy exercise in aerobic class. Very few documented cases of primary or spontaneous ASVT in absence of clear factors and in such anatomical location have been previously reported.</p></sec><sec id="Sec2"><title>Case presentation</title><p id="Par19">A 36 year old apparently healthy Hispanic female presented to the emergency department (ED) with a 4 days history of left upper extremity pain dull aching in nature and tenderness to palpation after a week of strenuous activity. Her background history was without any significant family history or risk factors. Physical examination revealed a moderately nourished, well-built female, not in acute distress except for marked pain in left extremity. No other abnormality was detected on physical examination.</p><p id="Par20">A complete blood count was done as part of a routine examination. Doppler studies of the four extremities was done in ED which showed left axillosubclavian acute DVT. Laboratory results are presented in Table <xref rid="Tab2" ref-type="table">2</xref> and Table <xref rid="Tab3" ref-type="table">3</xref> below. Secondary to the elevated D-Dimers patient underwent CT chest and pulmonary angiography to rule out pulmonary extension or pulmonary embolism (PE). The CT results confirmed the presence of left axillo-subclavian venous thrombosis; however, there was no evidence of PE.<table-wrap id="Tab2"><label>Table 2</label><caption><p>Initial Admission Laboratory Results</p></caption><table frame="hsides" rules="groups"><thead><tr><th>Parameter</th><th>Values</th><th>Range</th></tr></thead><tbody><tr><td>WBC</td><td>6.2</td><td>4–11</td></tr><tr><td>RBC</td><td>3.63</td><td>3.8–5.3</td></tr><tr><td>HCT</td><td>32</td><td>37–51</td></tr><tr><td>MCV</td><td>88.1</td><td>80–101</td></tr><tr><td>MCHC</td><td>30.8</td><td>27–32</td></tr><tr><td>RDW</td><td>12.6</td><td>11.5–14.5</td></tr><tr><td>Platelet</td><td>244</td><td>130–450</td></tr><tr><td>D-dimer</td><td>2203</td><td>0–278</td></tr><tr><td>BUN</td><td>15</td><td>8–25</td></tr><tr><td>Creatinine</td><td>0.7</td><td>0.5–1.04</td></tr><tr><td>Sodiun</td><td>138</td><td>133–145</td></tr><tr><td>Potassium</td><td>4.4</td><td>3.5–5.5</td></tr><tr><td>Chloride</td><td>101</td><td>96–101</td></tr><tr><td>Carbon dioxide</td><td>27</td><td>22–28</td></tr><tr><td>Calcium</td><td>9.4</td><td>8.5–10.5</td></tr><tr><td>Antithrombin III</td><td>113</td><td>85–120</td></tr><tr><td>Protein C</td><td>110</td><td>70–180</td></tr><tr><td>Lupus anticoagulant</td><td>45</td><td><40</td></tr><tr><td>Proteins</td><td>94</td><td>60–140</td></tr></tbody></table></table-wrap><table-wrap id="Tab3"><label>Table 3</label><caption><p>Risk factors for Paget-Schroetter Syndrome</p></caption><table frame="hsides" rules="groups"><thead><tr><th>Risk Factors</th><th></th></tr></thead><tbody><tr><td>Physical activity involving hyperabduction of the shoulder, as seen in weight lifters</td><td></td></tr><tr><td>Motions often associated with tennis players and baseball pitchers</td><td></td></tr><tr><td>Vigorous exercise of the neck and upper extremity muscles</td><td></td></tr><tr><td>Overdeveloped anterior scalene muscle Rudimentary first rib</td><td></td></tr><tr><td>Presence of cervical rib</td><td></td></tr><tr><td>Congenital band between first and second ribs Fracture of the clavicle with callus formation</td><td></td></tr><tr><td>Apical tumors of the superior sulcus of the lung (Pancoast tumor)</td><td></td></tr><tr><td>Thoracic outlet syndrome</td><td></td></tr></tbody></table></table-wrap></p><p id="Par21">Futhermore, a CT chest was done and results showed there were no anatomical abnormalities obstructing thoracic outlet. It is possible that strenuous physical activity with temporary obstruction of the thoracic outlet while patient was training her upper body has triggered and likely temporary dehydration caused by extensive sweating during physical training further contributed to the thrombotic event.</p><p id="Par22">The patient was started on strict precautions of left upper extremity immobilization, analgesics in the form of Tylenol 650 mg every 6 h for pain as well as cold compresses. Lovenox 90 mg subcutaneous twice daily (1 mg/kg BID) was started together with warfarin to keep INR 2–3. On the third day of hospitalization the therapeutic INR was reached and patient was discharged.</p><p id="Par23">Additional workup to exclude hypercoagulable state in the form of antiphospholilpid antibody, factor V, Leyden, protein S and C and antithrombin III were within normal levels with no gross abnormality suggestive of thrombophilic state. Catheter-guided thrombolysis was considered with option to transfer patient to specialized center since this type of treatment was not available at the described facility. However patient was not willing to relocate and preferred to be treated at the same facility she was admitted to originally knowing that other type of treatment is available at the other center.</p><p id="Par24">Two months after discharge, patient came for follow up. Doppler study showed that there were no blood clots in axillosubclavian vessels and all blood work was within normal limits including D-Dimers of 177 and the patient clinically asymptomatic.</p></sec><sec id="Sec3"><title>Discussion and conclusions</title><p id="Par25">In this report, we have noticed a relatively uncommon presentation of DVT in the upper extremity in absence of any common risk factor.</p><p id="Par26">Stress thrombosis or primary ASVT syndrome can occur in apparently healthy individuals without Virchow’s triad or other thrombosis enhancing risk factors. How can thrombosis occur in patients without any apparent predisposition? The exact mechanism of this is not well understood, however, it may be related to minor thoracic inlet abnormalities together with strenuous physical activity which defies the state of stasis in Virchow’s triad. The abnormalities of the thoracic outlet are often bilateral and predispose to eventual thrombosis of both venous systems [<xref ref-type="bibr" rid="CR15">15</xref>]. In addition, chronic compression of the vein can cause perivenous fibrosis, which may result in partial venous obstruction despite surgical correction of the compressing lesion [<xref ref-type="bibr" rid="CR15">15</xref>, <xref ref-type="bibr" rid="CR16">16</xref>]. In our case 2 months after hospitalization patient treated with anticoagulation with Warfarin came for a follow up. Her symptoms were resolved so to were blood clot in axillosubclavian veins.</p><p id="Par27">Condition management should include differential diagnosis such as cellulitis, lymphedema, neoplastic compression of veins, traumatic muscle injury, and thrombosis of superficial veins. A detailed imaging panel including dopplers, CT angiography and even MRI/MRA must be considered. Laboratory testing should include CBC with special focus on platelet count to exclude other etiologies, as well as a complete panel of hypercoagulable work up to exclude secondary causes [<xref ref-type="bibr" rid="CR17">17</xref>]. Should clinical picture suggest ASVT, the best test to perform first is the duplex sonogram. It is inexpensive, highly sensitive, specific and non-invasive way to diagnose condition without delay of treatment.</p><p id="Par28">Patients may have good prognosis and better outcome of thrombolytic therapy, if ASVT was diagnosed early, and extent of damages is limited. In case of skeletal abnormality, compressing venous structures as a cause of ASVT, surgical intervention such as rib or clavicle resection may be needed. Rehabilitation and physical therapy are an important part of patient’s management, even more so in case of purely muscular causes of venous blood flow obstruction.</p><p id="Par29">Acute deep venous thrombosis is a very common problem affecting up to one in every thousand Americans; however, upper extremity presentation is much less common. Excluding surgical causes, catheter-induced upper extremity venous thrombosis becomes a rare presentation. However by further excluding secondary causes only very few cases of primary upper extremity DVT or PSS have been reported. As a result, more research is needed in ASVT. Presently, there is a lack of guided management of rare conditions such as our case, or consensus among thesources. Physicians should be aware of this rare disease since untreated conditions may be debilitating for the patient and very costly especially if diagnosed with a delay. Absence of anatomical obstruction and favorable outcome after conventional treatment makes this case unique.</p></sec></body><back><glossary><title>Abbreviations</title><def-list><def-item><term>ASVT</term><def><p id="Par5">Axillary-subclavian venous thrombosis</p></def></def-item><def-item><term>CBC</term><def><p id="Par6">Complete Blood Clot</p></def></def-item><def-item><term>DVT</term><def><p id="Par7">Deep Vein Thrombosis</p></def></def-item><def-item><term>ED</term><def><p id="Par8">Emergency department</p></def></def-item><def-item><term>INR</term><def><p id="Par9">International Normalised Ratio</p></def></def-item><def-item><term>MRA</term><def><p id="Par10">Magnetic Resonance Angiogram</p></def></def-item><def-item><term>MRI</term><def><p id="Par11">Magnetic Resonance Imaging</p></def></def-item><def-item><term>PE</term><def><p id="Par12">Pulmonary embolism</p></def></def-item><def-item><term>PSS</term><def><p id="Par13">Paget-Schrotter Syndrome</p></def></def-item><def-item><term>UEDVT</term><def><p id="Par14">Upper Extremity Deep Vein Thrombosis</p></def></def-item></def-list></glossary><ack><title>Acknowledgements</title><p>We thank everyone involved for their kind support during this work.</p><sec id="FPar1"><title>Availability of data and materials</title><p id="Par30">Please contact author for data requests.</p></sec><sec id="FPar2"><title>Funding</title><p id="Par31">None.</p></sec></ack><notes notes-type="author-contribution"><title>Authors’ contributions</title><p>RI, ID, NA, AG worked on interpreting and analyzing patient data as well as writing of the article. MW, AK and RP worked on writing the article and critical revision of the article. All authors were major contributors. All the authors of this paper have reviewed the document in its entirety and are in agreement with the structure and content. All authors read and approved the final manuscript.</p></notes><notes notes-type="COI-statement"><sec id="FPar3"><title>Ethics approval and consent to participate</title><p id="Par32">Patient gave her consent to be in this study/case report.</p></sec><sec id="FPar4"><title>Consent for publication</title><p id="Par33">The patient gave her consent for the publication of this study.</p></sec><sec id="FPar5"><title>Competing interests</title><p id="Par34">The authors declare that they have no competing interests.</p></sec><sec id="FPar6"><title>Publisher’s Note</title><p id="Par35">Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.</p></sec></notes><ref-list id="Bib1"><title>References</title><ref id="CR1"><label>1.</label><mixed-citation publication-type="other">Dep A, et al. Paget-Schrotter syndrome and complications of management. 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