Nodular Proliferation in Parkes Weber Syndrome.
Identifieur interne : 000646 ( PubMed/Corpus ); précédent : 000645; suivant : 000647Nodular Proliferation in Parkes Weber Syndrome.
Auteurs : Diego José Rojas Esquivel ; Elena Marín Manzano ; Nieves Aleicel Concepci N ; Juan Carlos Lopéz Gutierréz ; Luis Riera De CubasSource :
- Annals of vascular surgery [ 1615-5947 ] ; 2017.
English descriptors
- KwdEn :
- Adult, Antibodies, Monoclonal, Murine-Derived, Antigens, CD31 (analysis), Antigens, CD34 (immunology), Arteries (chemistry), Arteries (diagnostic imaging), Arteries (pathology), Arteries (surgery), Arteriovenous Malformations (diagnostic imaging), Arteriovenous Malformations (pathology), Arteriovenous Malformations (surgery), Biopsy, Cell Proliferation, Computed Tomography Angiography, Humans, Immunohistochemistry, Lower Extremity (blood supply), Male, Sturge-Weber Syndrome (diagnostic imaging), Sturge-Weber Syndrome (pathology), Sturge-Weber Syndrome (surgery), Treatment Outcome, Vascular Surgical Procedures, Veins (chemistry), Veins (diagnostic imaging), Veins (pathology), Veins (surgery).
- MESH :
- chemical , analysis : Antigens, CD31.
- chemical , immunology : Antigens, CD34.
- chemical : Antibodies, Monoclonal, Murine-Derived.
- blood supply : Lower Extremity.
- chemistry : Arteries, Veins.
- diagnostic imaging : Arteries, Arteriovenous Malformations, Sturge-Weber Syndrome, Veins.
- pathology : Arteries, Arteriovenous Malformations, Sturge-Weber Syndrome, Veins.
- surgery : Arteries, Arteriovenous Malformations, Sturge-Weber Syndrome, Veins.
- Adult, Biopsy, Cell Proliferation, Computed Tomography Angiography, Humans, Immunohistochemistry, Male, Treatment Outcome, Vascular Surgical Procedures.
Abstract
Parkes Weber syndrome (PWS) is characterized by the association of high flow vascular malformation and overgrowth of a part of the body, usually a limb. In a previous review of 10 patients with PWS from our hospital we described a case of congenital short femur and four cases of severe lymphedema. We present a case of PWS associated with a nodular proliferative form not previously described. A 38 year old male with diagnosis of PWS with involvement of the right lower limb (RLL) was derived to our clinic. He complained about the appearance of painful nodular tumors in his RLL and some episodes of bleeding through the tumors. The physical examination revealed increased size of the RLL compared to left lower limb. Two nodular tumors were evident in his RLL. One located proximal in the leg and another one in ankle. The computed tomographic angiography revealed multiple arterio-venous shunts in the RLL. The tumors were not arterio-venous shunts, neither aneurysms. We decided to make surgical resection of the tumors. In the pathology analysis the tumors were positive for CD31, CD34 and negative for D240 markers. Eight months after surgery the patient had no recurrence of the tumors, and he is asymptomatic.The presence of nodular tumors in PWS has not been previously described. This makes us to think that these could be hamartomatous lesions similar to those of the CLOVES syndrome or a PIK3CA mutation.
DOI: 10.1016/j.avsg.2016.06.015
PubMed: 27575310
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Electronic address: doctorojas@gmail.com.</nlm:affiliation></affiliation></author><author><name sortKey="Marin Manzano, Elena" sort="Marin Manzano, Elena" uniqKey="Marin Manzano E" first="Elena" last="Marín Manzano">Elena Marín Manzano</name><affiliation><nlm:affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author><author><name sortKey="Concepci N, Nieves Aleicel" sort="Concepci N, Nieves Aleicel" uniqKey="Concepci N N" first="Nieves Aleicel" last="Concepci N">Nieves Aleicel Concepci N</name><affiliation><nlm:affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author><author><name sortKey="Lopez Gutierrez, Juan Carlos" sort="Lopez Gutierrez, Juan Carlos" uniqKey="Lopez Gutierrez J" first="Juan Carlos" last="Lopéz Gutierréz">Juan Carlos Lopéz Gutierréz</name><affiliation><nlm:affiliation>Department of Pediatric Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author><author><name sortKey="Riera De Cubas, Luis" sort="Riera De Cubas, Luis" uniqKey="Riera De Cubas L" first="Luis" last="Riera De Cubas">Luis Riera De Cubas</name><affiliation><nlm:affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2017">2017</date><idno type="RBID">pubmed:27575310</idno><idno type="pmid">27575310</idno><idno type="doi">10.1016/j.avsg.2016.06.015</idno><idno type="wicri:Area/PubMed/Corpus">000646</idno><idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Corpus" wicri:corpus="PubMed">000646</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Nodular Proliferation in Parkes Weber Syndrome.</title><author><name sortKey="Rojas Esquivel, Diego Jose" sort="Rojas Esquivel, Diego Jose" uniqKey="Rojas Esquivel D" first="Diego José" last="Rojas Esquivel">Diego José Rojas Esquivel</name><affiliation><nlm:affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain. Electronic address: doctorojas@gmail.com.</nlm:affiliation></affiliation></author><author><name sortKey="Marin Manzano, Elena" sort="Marin Manzano, Elena" uniqKey="Marin Manzano E" first="Elena" last="Marín Manzano">Elena Marín Manzano</name><affiliation><nlm:affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author><author><name sortKey="Concepci N, Nieves Aleicel" sort="Concepci N, Nieves Aleicel" uniqKey="Concepci N N" first="Nieves Aleicel" last="Concepci N">Nieves Aleicel Concepci N</name><affiliation><nlm:affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author><author><name sortKey="Lopez Gutierrez, Juan Carlos" sort="Lopez Gutierrez, Juan Carlos" uniqKey="Lopez Gutierrez J" first="Juan Carlos" last="Lopéz Gutierréz">Juan Carlos Lopéz Gutierréz</name><affiliation><nlm:affiliation>Department of Pediatric Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author><author><name sortKey="Riera De Cubas, Luis" sort="Riera De Cubas, Luis" uniqKey="Riera De Cubas L" first="Luis" last="Riera De Cubas">Luis Riera De Cubas</name><affiliation><nlm:affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</nlm:affiliation></affiliation></author></analytic><series><title level="j">Annals of vascular surgery</title><idno type="eISSN">1615-5947</idno><imprint><date when="2017" type="published">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Antibodies, Monoclonal, Murine-Derived</term><term>Antigens, CD31 (analysis)</term><term>Antigens, CD34 (immunology)</term><term>Arteries (chemistry)</term><term>Arteries (diagnostic imaging)</term><term>Arteries (pathology)</term><term>Arteries (surgery)</term><term>Arteriovenous Malformations (diagnostic imaging)</term><term>Arteriovenous Malformations (pathology)</term><term>Arteriovenous Malformations (surgery)</term><term>Biopsy</term><term>Cell Proliferation</term><term>Computed Tomography Angiography</term><term>Humans</term><term>Immunohistochemistry</term><term>Lower Extremity (blood supply)</term><term>Male</term><term>Sturge-Weber Syndrome (diagnostic imaging)</term><term>Sturge-Weber Syndrome (pathology)</term><term>Sturge-Weber Syndrome (surgery)</term><term>Treatment Outcome</term><term>Vascular Surgical Procedures</term><term>Veins (chemistry)</term><term>Veins (diagnostic imaging)</term><term>Veins (pathology)</term><term>Veins (surgery)</term></keywords><keywords scheme="MESH" type="chemical" qualifier="analysis" xml:lang="en"><term>Antigens, CD31</term></keywords><keywords scheme="MESH" type="chemical" qualifier="immunology" xml:lang="en"><term>Antigens, CD34</term></keywords><keywords scheme="MESH" type="chemical" xml:lang="en"><term>Antibodies, Monoclonal, Murine-Derived</term></keywords><keywords scheme="MESH" qualifier="blood supply" xml:lang="en"><term>Lower Extremity</term></keywords><keywords scheme="MESH" qualifier="chemistry" xml:lang="en"><term>Arteries</term><term>Veins</term></keywords><keywords scheme="MESH" qualifier="diagnostic imaging" xml:lang="en"><term>Arteries</term><term>Arteriovenous Malformations</term><term>Sturge-Weber Syndrome</term><term>Veins</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Arteries</term><term>Arteriovenous Malformations</term><term>Sturge-Weber Syndrome</term><term>Veins</term></keywords><keywords scheme="MESH" qualifier="surgery" xml:lang="en"><term>Arteries</term><term>Arteriovenous Malformations</term><term>Sturge-Weber Syndrome</term><term>Veins</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Biopsy</term><term>Cell Proliferation</term><term>Computed Tomography Angiography</term><term>Humans</term><term>Immunohistochemistry</term><term>Male</term><term>Treatment Outcome</term><term>Vascular Surgical Procedures</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Parkes Weber syndrome (PWS) is characterized by the association of high flow vascular malformation and overgrowth of a part of the body, usually a limb. In a previous review of 10 patients with PWS from our hospital we described a case of congenital short femur and four cases of severe lymphedema. We present a case of PWS associated with a nodular proliferative form not previously described. A 38 year old male with diagnosis of PWS with involvement of the right lower limb (RLL) was derived to our clinic. He complained about the appearance of painful nodular tumors in his RLL and some episodes of bleeding through the tumors. The physical examination revealed increased size of the RLL compared to left lower limb. Two nodular tumors were evident in his RLL. One located proximal in the leg and another one in ankle. The computed tomographic angiography revealed multiple arterio-venous shunts in the RLL. The tumors were not arterio-venous shunts, neither aneurysms. We decided to make surgical resection of the tumors. In the pathology analysis the tumors were positive for CD31, CD34 and negative for D240 markers. Eight months after surgery the patient had no recurrence of the tumors, and he is asymptomatic.The presence of nodular tumors in PWS has not been previously described. This makes us to think that these could be hamartomatous lesions similar to those of the CLOVES syndrome or a PIK3CA mutation.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">27575310</PMID><DateCreated><Year>2016</Year><Month>08</Month><Day>30</Day></DateCreated><DateCompleted><Year>2017</Year><Month>01</Month><Day>23</Day></DateCompleted><DateRevised><Year>2017</Year><Month>01</Month><Day>23</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1615-5947</ISSN><JournalIssue CitedMedium="Internet"><Volume>38</Volume><PubDate><Year>2017</Year><Month>Jan</Month></PubDate></JournalIssue><Title>Annals of vascular surgery</Title><ISOAbbreviation>Ann Vasc Surg</ISOAbbreviation></Journal><ArticleTitle>Nodular Proliferation in Parkes Weber Syndrome.</ArticleTitle><Pagination><MedlinePgn>321.e1-321.e4</MedlinePgn></Pagination><ELocationID EIdType="pii" ValidYN="Y">S0890-5096(16)30712-9</ELocationID><ELocationID EIdType="doi" ValidYN="Y">10.1016/j.avsg.2016.06.015</ELocationID><Abstract><AbstractText>Parkes Weber syndrome (PWS) is characterized by the association of high flow vascular malformation and overgrowth of a part of the body, usually a limb. In a previous review of 10 patients with PWS from our hospital we described a case of congenital short femur and four cases of severe lymphedema. We present a case of PWS associated with a nodular proliferative form not previously described. A 38 year old male with diagnosis of PWS with involvement of the right lower limb (RLL) was derived to our clinic. He complained about the appearance of painful nodular tumors in his RLL and some episodes of bleeding through the tumors. The physical examination revealed increased size of the RLL compared to left lower limb. Two nodular tumors were evident in his RLL. One located proximal in the leg and another one in ankle. The computed tomographic angiography revealed multiple arterio-venous shunts in the RLL. The tumors were not arterio-venous shunts, neither aneurysms. We decided to make surgical resection of the tumors. In the pathology analysis the tumors were positive for CD31, CD34 and negative for D240 markers. Eight months after surgery the patient had no recurrence of the tumors, and he is asymptomatic.The presence of nodular tumors in PWS has not been previously described. This makes us to think that these could be hamartomatous lesions similar to those of the CLOVES syndrome or a PIK3CA mutation.</AbstractText><CopyrightInformation>Copyright © 2016 Elsevier Inc. All rights reserved.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Rojas Esquivel</LastName><ForeName>Diego José</ForeName><Initials>DJ</Initials><AffiliationInfo><Affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain. Electronic address: doctorojas@gmail.com.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Marín Manzano</LastName><ForeName>Elena</ForeName><Initials>E</Initials><AffiliationInfo><Affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Concepción</LastName><ForeName>Nieves Aleicel</ForeName><Initials>NA</Initials><AffiliationInfo><Affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Lopéz Gutierréz</LastName><ForeName>Juan Carlos</ForeName><Initials>JC</Initials><AffiliationInfo><Affiliation>Department of Pediatric Surgery, La Paz University Hospital, Madrid, Spain.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Riera de Cubas</LastName><ForeName>Luis</ForeName><Initials>L</Initials><AffiliationInfo><Affiliation>Department of Vascular Surgery, La Paz University Hospital, Madrid, Spain.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D002363">Case Reports</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2016</Year><Month>08</Month><Day>26</Day></ArticleDate></Article><MedlineJournalInfo><Country>Netherlands</Country><MedlineTA>Ann Vasc Surg</MedlineTA><NlmUniqueID>8703941</NlmUniqueID><ISSNLinking>0890-5096</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D058846">Antibodies, Monoclonal, Murine-Derived</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D019408">Antigens, CD31</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D018952">Antigens, CD34</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="C500987">monoclonal antibody D2-40</NameOfSubstance></Chemical></ChemicalList><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName UI="D000328" MajorTopicYN="N">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D058846" MajorTopicYN="N">Antibodies, Monoclonal, Murine-Derived</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D019408" MajorTopicYN="N">Antigens, CD31</DescriptorName><QualifierName UI="Q000032" MajorTopicYN="N">analysis</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D018952" MajorTopicYN="N">Antigens, CD34</DescriptorName><QualifierName UI="Q000276" MajorTopicYN="N">immunology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D001158" MajorTopicYN="N">Arteries</DescriptorName><QualifierName UI="Q000737" MajorTopicYN="N">chemistry</QualifierName><QualifierName UI="Q000000981" MajorTopicYN="N">diagnostic imaging</QualifierName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName><QualifierName UI="Q000601" MajorTopicYN="N">surgery</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D001165" MajorTopicYN="N">Arteriovenous Malformations</DescriptorName><QualifierName UI="Q000000981" MajorTopicYN="N">diagnostic imaging</QualifierName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName><QualifierName UI="Q000601" MajorTopicYN="N">surgery</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D001706" MajorTopicYN="N">Biopsy</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D049109" MajorTopicYN="Y">Cell Proliferation</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D000072226" MajorTopicYN="N">Computed Tomography Angiography</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D007150" MajorTopicYN="N">Immunohistochemistry</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D035002" MajorTopicYN="N">Lower Extremity</DescriptorName><QualifierName UI="Q000098" MajorTopicYN="Y">blood supply</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D008297" MajorTopicYN="N">Male</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D013341" MajorTopicYN="N">Sturge-Weber Syndrome</DescriptorName><QualifierName UI="Q000000981" MajorTopicYN="N">diagnostic imaging</QualifierName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName><QualifierName UI="Q000601" MajorTopicYN="N">surgery</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D016896" MajorTopicYN="N">Treatment Outcome</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D014656" MajorTopicYN="N">Vascular Surgical Procedures</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D014680" MajorTopicYN="N">Veins</DescriptorName><QualifierName UI="Q000737" MajorTopicYN="N">chemistry</QualifierName><QualifierName UI="Q000000981" MajorTopicYN="N">diagnostic imaging</QualifierName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName><QualifierName UI="Q000601" MajorTopicYN="N">surgery</QualifierName></MeshHeading></MeshHeadingList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2016</Year><Month>02</Month><Day>05</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2016</Year><Month>06</Month><Day>05</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2016</Year><Month>06</Month><Day>12</Day></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2016</Year><Month>8</Month><Day>31</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2017</Year><Month>1</Month><Day>24</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2016</Year><Month>8</Month><Day>31</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">27575310</ArticleId><ArticleId IdType="pii">S0890-5096(16)30712-9</ArticleId><ArticleId IdType="doi">10.1016/j.avsg.2016.06.015</ArticleId></ArticleIdList></PubmedData></pubmed></record>Pour manipuler ce document sous Unix (Dilib)
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