Generalized lymphedema associated with neurologic signs (GLANS) syndrome: a new entity?
Identifieur interne : 001150 ( PubMed/Curation ); précédent : 001149; suivant : 001151Generalized lymphedema associated with neurologic signs (GLANS) syndrome: a new entity?
Auteurs : Marine Berton [France] ; Gérard Lorette [France] ; Françoise Baulieu [France] ; Emmanuelle Lagrue [France] ; Sophie Blesson [France] ; Frédéric Cambazard [France] ; Loïc Vaillant [France] ; Annabel Maruani [France]Source :
- Journal of the American Academy of Dermatology [ 1097-6787 ] ; 2015.
Descripteurs français
- KwdFr :
- MESH :
English descriptors
- KwdEn :
- Adolescent, Adult, Child, Electroencephalography, Epilepsy (diagnosis), Extremities, Face, Fatal Outcome, Female, Humans, Lymphedema (congenital), Lymphedema (diagnosis), Lymphedema (therapy), Male, Nervous System Diseases (diagnosis), Neuroimaging, Sleep Wake Disorders (diagnosis), Tomography, X-Ray Computed.
- MESH :
- congenital : Lymphedema.
- diagnosis : Epilepsy, Lymphedema, Nervous System Diseases, Sleep Wake Disorders.
- therapy : Lymphedema.
- Adolescent, Adult, Child, Electroencephalography, Extremities, Face, Fatal Outcome, Female, Humans, Male, Neuroimaging, Tomography, X-Ray Computed.
Abstract
Primary lymphedema in children, especially generalized disease with facial involvement, is rare.
DOI: 10.1016/j.jaad.2014.10.033
PubMed: 25484267
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Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours</wicri:regionArea></affiliation></author><author><name sortKey="Lorette, Gerard" sort="Lorette, Gerard" uniqKey="Lorette G" first="Gérard" last="Lorette">Gérard Lorette</name><affiliation wicri:level="1"><nlm:affiliation>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours</wicri:regionArea></affiliation></author><author><name sortKey="Baulieu, Francoise" sort="Baulieu, Francoise" uniqKey="Baulieu F" first="Françoise" last="Baulieu">Françoise Baulieu</name><affiliation wicri:level="1"><nlm:affiliation>University François Rabelais, Tours, France; Department of Nuclear Medicine, Centre Hospitalier Régional Universitaire, Tours, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>University François Rabelais, Tours, France; Department of Nuclear Medicine, Centre Hospitalier Régional Universitaire, Tours</wicri:regionArea></affiliation></author><author><name sortKey="Lagrue, Emmanuelle" sort="Lagrue, Emmanuelle" uniqKey="Lagrue E" first="Emmanuelle" last="Lagrue">Emmanuelle Lagrue</name><affiliation wicri:level="1"><nlm:affiliation>University François Rabelais, Tours, France; Department of Pediatric Neurology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>University François Rabelais, Tours, France; Department of Pediatric Neurology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours</wicri:regionArea></affiliation></author><author><name sortKey="Blesson, Sophie" sort="Blesson, Sophie" uniqKey="Blesson S" first="Sophie" last="Blesson">Sophie Blesson</name><affiliation wicri:level="1"><nlm:affiliation>Department of Genetics, Centre Hospitalier Régional Universitaire, Tours, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>Department of Genetics, Centre Hospitalier Régional Universitaire, Tours</wicri:regionArea></affiliation></author><author><name sortKey="Cambazard, Frederic" sort="Cambazard, Frederic" uniqKey="Cambazard F" first="Frédéric" last="Cambazard">Frédéric Cambazard</name><affiliation wicri:level="1"><nlm:affiliation>Department of Dermatology, University Hospital of Saint-Etienne, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, University Hospital of Saint-Etienne</wicri:regionArea></affiliation></author><author><name sortKey="Vaillant, Loic" sort="Vaillant, Loic" uniqKey="Vaillant L" first="Loïc" last="Vaillant">Loïc Vaillant</name><affiliation wicri:level="1"><nlm:affiliation>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours</wicri:regionArea></affiliation></author><author><name sortKey="Maruani, Annabel" sort="Maruani, Annabel" uniqKey="Maruani A" first="Annabel" last="Maruani">Annabel Maruani</name><affiliation wicri:level="1"><nlm:affiliation>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours, France. Electronic address: annabel.maruani@univ-tours.fr.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours</wicri:regionArea></affiliation></author></analytic><series><title level="j">Journal of the American Academy of Dermatology</title><idno type="eISSN">1097-6787</idno><imprint><date when="2015" type="published">2015</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent</term><term>Adult</term><term>Child</term><term>Electroencephalography</term><term>Epilepsy (diagnosis)</term><term>Extremities</term><term>Face</term><term>Fatal Outcome</term><term>Female</term><term>Humans</term><term>Lymphedema (congenital)</term><term>Lymphedema (diagnosis)</term><term>Lymphedema (therapy)</term><term>Male</term><term>Nervous System Diseases (diagnosis)</term><term>Neuroimaging</term><term>Sleep Wake Disorders (diagnosis)</term><term>Tomography, X-Ray Computed</term></keywords><keywords scheme="KwdFr" xml:lang="fr"><term>Adolescent</term><term>Adulte</term><term>Enfant</term><term>Face</term><term>Femelle</term><term>Humains</term><term>Issue fatale</term><term>Lymphoedème ()</term><term>Lymphoedème (diagnostic)</term><term>Maladies du système nerveux (diagnostic)</term><term>Membres</term><term>Mâle</term><term>Neuroimagerie</term><term>Tomodensitométrie</term><term>Troubles de la veille et du sommeil (diagnostic)</term><term>Électroencéphalographie</term><term>Épilepsie (diagnostic)</term></keywords><keywords scheme="MESH" qualifier="congenital" xml:lang="en"><term>Lymphedema</term></keywords><keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Epilepsy</term><term>Lymphedema</term><term>Nervous System Diseases</term><term>Sleep Wake Disorders</term></keywords><keywords scheme="MESH" qualifier="diagnostic" xml:lang="fr"><term>Lymphoedème</term><term>Maladies du système nerveux</term><term>Troubles de la veille et du sommeil</term><term>Épilepsie</term></keywords><keywords scheme="MESH" qualifier="therapy" xml:lang="en"><term>Lymphedema</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Adult</term><term>Child</term><term>Electroencephalography</term><term>Extremities</term><term>Face</term><term>Fatal Outcome</term><term>Female</term><term>Humans</term><term>Male</term><term>Neuroimaging</term><term>Tomography, X-Ray Computed</term></keywords><keywords scheme="MESH" xml:lang="fr"><term>Adolescent</term><term>Adulte</term><term>Enfant</term><term>Face</term><term>Femelle</term><term>Humains</term><term>Issue fatale</term><term>Lymphoedème</term><term>Membres</term><term>Mâle</term><term>Neuroimagerie</term><term>Tomodensitométrie</term><term>Électroencéphalographie</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Primary lymphedema in children, especially generalized disease with facial involvement, is rare.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">25484267</PMID><DateCreated><Year>2015</Year><Month>01</Month><Day>16</Day></DateCreated><DateCompleted><Year>2015</Year><Month>03</Month><Day>11</Day></DateCompleted><DateRevised><Year>2015</Year><Month>11</Month><Day>19</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1097-6787</ISSN><JournalIssue CitedMedium="Internet"><Volume>72</Volume><Issue>2</Issue><PubDate><Year>2015</Year><Month>Feb</Month></PubDate></JournalIssue><Title>Journal of the American Academy of Dermatology</Title><ISOAbbreviation>J. Am. Acad. Dermatol.</ISOAbbreviation></Journal><ArticleTitle>Generalized lymphedema associated with neurologic signs (GLANS) syndrome: a new entity?</ArticleTitle><Pagination><MedlinePgn>333-9</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1016/j.jaad.2014.10.033</ELocationID><ELocationID EIdType="pii" ValidYN="Y">S0190-9622(14)02110-0</ELocationID><Abstract><AbstractText Label="BACKGROUND" NlmCategory="BACKGROUND">Primary lymphedema in children, especially generalized disease with facial involvement, is rare.</AbstractText><AbstractText Label="OBJECTIVE" NlmCategory="OBJECTIVE">We sought to report 3 childhood cases of lymphedema with associated neurologic findings and to provide a pathophysiologic explanation for this association.</AbstractText><AbstractText Label="METHODS" NlmCategory="METHODS">Clinical observations, electroencephalography, and neuroimaging studies were evaluated. Microcomparative genomic hybridization was performed in 1 case.</AbstractText><AbstractText Label="RESULTS" NlmCategory="RESULTS">The 3 children had primary lymphedema of all 4 limbs and the face. This was confirmed by lymphoscintigraphy, which showed hypoplasia of vessels and hypofixation of lymph nodes. They had nonspecific neurologic disorders and electroencephalography abnormalities, without intellectual deficit. Neuroimaging revealed normal findings. Microcomparative genomic hybridization in 1 patient revealed no cytogenetic anomaly. The outcome was fatal in 1 case with development of visceral lymphedema and coma.</AbstractText><AbstractText Label="LIMITATIONS" NlmCategory="CONCLUSIONS">Genetic studies were performed in only 1 case.</AbstractText><AbstractText Label="CONCLUSION" NlmCategory="CONCLUSIONS">These observations suggest that neurologic assessment and electroencephalography are indicated for patients with lymphedema of the limbs and face to identify this syndrome.</AbstractText><CopyrightInformation>Copyright © 2014 American Academy of Dermatology, Inc. Published by Elsevier Inc. All rights reserved.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Berton</LastName><ForeName>Marine</ForeName><Initials>M</Initials><AffiliationInfo><Affiliation>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Lorette</LastName><ForeName>Gérard</ForeName><Initials>G</Initials><AffiliationInfo><Affiliation>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Baulieu</LastName><ForeName>Françoise</ForeName><Initials>F</Initials><AffiliationInfo><Affiliation>University François Rabelais, Tours, France; Department of Nuclear Medicine, Centre Hospitalier Régional Universitaire, Tours, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Lagrue</LastName><ForeName>Emmanuelle</ForeName><Initials>E</Initials><AffiliationInfo><Affiliation>University François Rabelais, Tours, France; Department of Pediatric Neurology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Blesson</LastName><ForeName>Sophie</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>Department of Genetics, Centre Hospitalier Régional Universitaire, Tours, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Cambazard</LastName><ForeName>Frédéric</ForeName><Initials>F</Initials><AffiliationInfo><Affiliation>Department of Dermatology, University Hospital of Saint-Etienne, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Vaillant</LastName><ForeName>Loïc</ForeName><Initials>L</Initials><AffiliationInfo><Affiliation>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Maruani</LastName><ForeName>Annabel</ForeName><Initials>A</Initials><AffiliationInfo><Affiliation>University François Rabelais, Tours, France; Department of Dermatology, Centre Hospitalier Régional Universitaire, Tours, France; Inserm U930, Tours, France. Electronic address: annabel.maruani@univ-tours.fr.</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>2014</Year><Month>12</Month><Day>04</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>J Am Acad Dermatol</MedlineTA><NlmUniqueID>7907132</NlmUniqueID><ISSNLinking>0190-9622</ISSNLinking></MedlineJournalInfo><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName UI="D000293" MajorTopicYN="N">Adolescent</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D000328" MajorTopicYN="N">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D002648" MajorTopicYN="N">Child</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D004569" MajorTopicYN="N">Electroencephalography</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D004827" MajorTopicYN="N">Epilepsy</DescriptorName><QualifierName UI="Q000175" MajorTopicYN="Y">diagnosis</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D005121" MajorTopicYN="N">Extremities</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005145" MajorTopicYN="N">Face</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D017809" MajorTopicYN="N">Fatal Outcome</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D008209" MajorTopicYN="N">Lymphedema</DescriptorName><QualifierName UI="Q000151" MajorTopicYN="N">congenital</QualifierName><QualifierName UI="Q000175" MajorTopicYN="Y">diagnosis</QualifierName><QualifierName UI="Q000628" MajorTopicYN="N">therapy</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D008297" MajorTopicYN="N">Male</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D009422" MajorTopicYN="N">Nervous System Diseases</DescriptorName><QualifierName UI="Q000175" MajorTopicYN="N">diagnosis</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D059906" MajorTopicYN="N">Neuroimaging</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D012893" MajorTopicYN="N">Sleep Wake Disorders</DescriptorName><QualifierName UI="Q000175" MajorTopicYN="Y">diagnosis</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D014057" MajorTopicYN="N">Tomography, X-Ray Computed</DescriptorName></MeshHeading></MeshHeadingList><KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">epilepsy</Keyword><Keyword MajorTopicYN="N">lymphatic</Keyword><Keyword MajorTopicYN="N">lymphedema</Keyword><Keyword MajorTopicYN="N">neurology</Keyword><Keyword MajorTopicYN="N">primary lymphedema</Keyword><Keyword MajorTopicYN="N">seizure</Keyword></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2014</Year><Month>02</Month><Day>12</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2014</Year><Month>10</Month><Day>21</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2014</Year><Month>10</Month><Day>24</Day></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2014</Year><Month>12</Month><Day>9</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2014</Year><Month>12</Month><Day>9</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2015</Year><Month>3</Month><Day>12</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">25484267</ArticleId><ArticleId IdType="pii">S0190-9622(14)02110-0</ArticleId><ArticleId IdType="doi">10.1016/j.jaad.2014.10.033</ArticleId></ArticleIdList></PubmedData></pubmed></record>Pour manipuler ce document sous Unix (Dilib)
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