Servelle-Martorell syndrome with extensive upper limb involvement: a case report
Identifieur interne : 003888 ( Pmc/Checkpoint ); précédent : 003887; suivant : 003889Servelle-Martorell syndrome with extensive upper limb involvement: a case report
Auteurs : Raju Karuppal [Inde] ; Rajendran V. Raman [Inde] ; Brijesh P. Valsalan [Inde] ; Ts Gopakumar [Inde] ; Chathoth Meethal Kumaran [Inde] ; Chembu Kara Vasu [Inde]Source :
- Journal of Medical Case Reports [ 1752-1947 ] ; 2008.
Abstract
Angio-osteohypotrophic syndrome is also known as Servelle-Martorell angiodysplasia. It is characterized by venous or, rarely, arterial malformations, which may result in limb hypertrophy and bony hypoplasia. Extensive involvement of the upper limb is a rare feature of Servelle-Martorell syndrome. Cases with minimal upper limb involvement have been described in the literature.
A young man presented with multiple separate swollen areas over the right upper limb and functional difficulty since birth. The arm muscles and muscles of the limb girdle were atrophic. The forearm and hand bones were hypoplastic and tender.
We report a case of Servelle-Martorell syndrome with extensive involvement of the entire upper limb and periscapular region. Servelle-Martorell syndrome is highlighted as one of the causes of angiodysplastic limb hypertrophy.
Url:
DOI: 10.1186/1752-1947-2-142
PubMed: 18454870
PubMed Central: 2394530
Affiliations:
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Raman</name><affiliation wicri:level="1"><nlm:aff id="I1">Medical College, Calicut, 673008 Kerala, India</nlm:aff><country xml:lang="fr">Inde</country><wicri:regionArea>Medical College, Calicut, 673008 Kerala</wicri:regionArea><wicri:noRegion>673008 Kerala</wicri:noRegion></affiliation></author><author><name sortKey="Valsalan, Brijesh P" sort="Valsalan, Brijesh P" uniqKey="Valsalan B" first="Brijesh P" last="Valsalan">Brijesh P. Valsalan</name><affiliation wicri:level="1"><nlm:aff id="I1">Medical College, Calicut, 673008 Kerala, India</nlm:aff><country xml:lang="fr">Inde</country><wicri:regionArea>Medical College, Calicut, 673008 Kerala</wicri:regionArea><wicri:noRegion>673008 Kerala</wicri:noRegion></affiliation></author><author><name sortKey="Gopakumar, Ts" sort="Gopakumar, Ts" uniqKey="Gopakumar T" first="Ts" last="Gopakumar">Ts Gopakumar</name><affiliation wicri:level="1"><nlm:aff id="I1">Medical College, Calicut, 673008 Kerala, India</nlm:aff><country xml:lang="fr">Inde</country><wicri:regionArea>Medical College, Calicut, 673008 Kerala</wicri:regionArea><wicri:noRegion>673008 Kerala</wicri:noRegion></affiliation></author><author><name sortKey="Kumaran, Chathoth Meethal" sort="Kumaran, Chathoth Meethal" uniqKey="Kumaran C" first="Chathoth Meethal" last="Kumaran">Chathoth Meethal Kumaran</name><affiliation wicri:level="1"><nlm:aff id="I1">Medical College, Calicut, 673008 Kerala, India</nlm:aff><country xml:lang="fr">Inde</country><wicri:regionArea>Medical College, Calicut, 673008 Kerala</wicri:regionArea><wicri:noRegion>673008 Kerala</wicri:noRegion></affiliation></author><author><name sortKey="Vasu, Chembu Kara" sort="Vasu, Chembu Kara" uniqKey="Vasu C" first="Chembu Kara" last="Vasu">Chembu Kara Vasu</name><affiliation wicri:level="1"><nlm:aff id="I1">Medical College, Calicut, 673008 Kerala, India</nlm:aff><country xml:lang="fr">Inde</country><wicri:regionArea>Medical College, Calicut, 673008 Kerala</wicri:regionArea><wicri:noRegion>673008 Kerala</wicri:noRegion></affiliation></author></analytic><series><title level="j">Journal of Medical Case Reports</title><idno type="eISSN">1752-1947</idno><imprint><date when="2008">2008</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec><title>Introduction</title><p>Angio-osteohypotrophic syndrome is also known as Servelle-Martorell angiodysplasia. It is characterized by venous or, rarely, arterial malformations, which may result in limb hypertrophy and bony hypoplasia. Extensive involvement of the upper limb is a rare feature of Servelle-Martorell syndrome. Cases with minimal upper limb involvement have been described in the literature.</p></sec><sec><title>Case presentation</title><p>A young man presented with multiple separate swollen areas over the right upper limb and functional difficulty since birth. The arm muscles and muscles of the limb girdle were atrophic. The forearm and hand bones were hypoplastic and tender.</p></sec><sec><title>Conclusion</title><p>We report a case of Servelle-Martorell syndrome with extensive involvement of the entire upper limb and periscapular region. Servelle-Martorell syndrome is highlighted as one of the causes of angiodysplastic limb hypertrophy.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct><biblStruct></biblStruct></listBibl></div1></back></TEI><pmc article-type="research-article"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">J Med Case Reports</journal-id><journal-title>Journal of Medical Case Reports</journal-title><issn pub-type="epub">1752-1947</issn><publisher><publisher-name>BioMed Central</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">18454870</article-id><article-id pub-id-type="pmc">2394530</article-id><article-id pub-id-type="publisher-id">1752-1947-2-142</article-id><article-id pub-id-type="doi">10.1186/1752-1947-2-142</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Servelle-Martorell syndrome with extensive upper limb involvement: a case report</article-title></title-group><contrib-group><contrib id="A1" corresp="yes" contrib-type="author"><name><surname>Karuppal</surname><given-names>Raju</given-names></name><xref ref-type="aff" rid="I1">1</xref><email>drrajuortho@rediffmail.com</email></contrib><contrib id="A2" contrib-type="author"><name><surname>Raman</surname><given-names>Rajendran V</given-names></name><xref ref-type="aff" rid="I1">1</xref><email>rajendran@hotmail.com</email></contrib><contrib id="A3" contrib-type="author"><name><surname>Valsalan</surname><given-names>Brijesh P</given-names></name><xref ref-type="aff" rid="I1">1</xref><email>brijeshpv@gmail.com</email></contrib><contrib id="A4" contrib-type="author"><name><surname>Gopakumar</surname><given-names>TS</given-names></name><xref ref-type="aff" rid="I1">1</xref><email>gopats@sifymail.com</email></contrib><contrib id="A5" contrib-type="author"><name><surname>Kumaran</surname><given-names>Chathoth Meethal</given-names></name><xref ref-type="aff" rid="I1">1</xref><email>cmk5303@yahoo.co.in</email></contrib><contrib id="A6" contrib-type="author"><name><surname>Vasu</surname><given-names>Chembu Kara</given-names></name><xref ref-type="aff" rid="I1">1</xref><email>ckvasu2000@rediffmail.com</email></contrib></contrib-group><aff id="I1"><label>1</label>Medical College, Calicut, 673008 Kerala, India</aff><pub-date pub-type="collection"><year>2008</year></pub-date><pub-date pub-type="epub"><day>3</day><month>5</month><year>2008</year></pub-date><volume>2</volume><fpage>142</fpage><lpage>142</lpage><ext-link ext-link-type="uri" xlink:href="http://www.jmedicalcasereports.com/content/2/1/142"></ext-link><history><date date-type="received"><day>4</day><month>9</month><year>2007</year></date><date date-type="accepted"><day>3</day><month>5</month><year>2008</year></date></history><permissions><copyright-statement>Copyright © 2008 Karuppal et al; licensee BioMed Central Ltd.</copyright-statement><copyright-year>2008</copyright-year><copyright-holder>Karuppal et al; licensee BioMed Central Ltd.</copyright-holder><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/2.0"><p>This is an Open Access article distributed under the terms of the Creative Commons Attribution License (<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by/2.0"></ext-link>), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</p><pmc-comment>
Karuppal
Raju
drrajuortho@rediffmail.com
Servelle-Martorell syndrome with extensive upper limb involvement: a case report
2008 Journal of Medical Case Reports 2(1): 142-. (2008) 1752-1947(2008)2:1<142> urn:ISSN:1752-1947
</license></permissions><abstract><sec><title>Introduction</title><p>Angio-osteohypotrophic syndrome is also known as Servelle-Martorell angiodysplasia. It is characterized by venous or, rarely, arterial malformations, which may result in limb hypertrophy and bony hypoplasia. Extensive involvement of the upper limb is a rare feature of Servelle-Martorell syndrome. Cases with minimal upper limb involvement have been described in the literature.</p></sec><sec><title>Case presentation</title><p>A young man presented with multiple separate swollen areas over the right upper limb and functional difficulty since birth. The arm muscles and muscles of the limb girdle were atrophic. The forearm and hand bones were hypoplastic and tender.</p></sec><sec><title>Conclusion</title><p>We report a case of Servelle-Martorell syndrome with extensive involvement of the entire upper limb and periscapular region. Servelle-Martorell syndrome is highlighted as one of the causes of angiodysplastic limb hypertrophy.</p></sec></abstract></article-meta></front></pmc><affiliations><list><country><li>Inde</li></country></list><tree><country name="Inde"><noRegion><name sortKey="Karuppal, Raju" sort="Karuppal, Raju" uniqKey="Karuppal R" first="Raju" last="Karuppal">Raju Karuppal</name></noRegion><name sortKey="Gopakumar, Ts" sort="Gopakumar, Ts" uniqKey="Gopakumar T" first="Ts" last="Gopakumar">Ts Gopakumar</name><name sortKey="Kumaran, Chathoth Meethal" sort="Kumaran, Chathoth Meethal" uniqKey="Kumaran C" first="Chathoth Meethal" last="Kumaran">Chathoth Meethal Kumaran</name><name sortKey="Raman, Rajendran V" sort="Raman, Rajendran V" uniqKey="Raman R" first="Rajendran V" last="Raman">Rajendran V. Raman</name><name sortKey="Valsalan, Brijesh P" sort="Valsalan, Brijesh P" uniqKey="Valsalan B" first="Brijesh P" last="Valsalan">Brijesh P. Valsalan</name><name sortKey="Vasu, Chembu Kara" sort="Vasu, Chembu Kara" uniqKey="Vasu C" first="Chembu Kara" last="Vasu">Chembu Kara Vasu</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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