Treatment of EARLY‐ONSET Gorham Syndrome with 8‐Year Follow‐Up
Identifieur interne : 007093 ( Istex/Corpus ); précédent : 007092; suivant : 007094Treatment of EARLY‐ONSET Gorham Syndrome with 8‐Year Follow‐Up
Auteurs : Annabel Maruani ; Sylvie Thimon ; Benoît De Courtivron ; Gonzague De Pinieux ; Françoise Baulieu ; Laurent Machet ; Gérard LoretteSource :
- Pediatric Dermatology [ 0736-8046 ] ; 2013-05.
Abstract
Abstract: A case of Gorham disease with several years of follow‐up is reported. At birth he had a mass in the thigh which was had pathology demonstrating a lymphangioma. By age 3 years, he had lymphedema of the ipsilateral foot and discrepant leg lengths. Radiography revealed heterogenous dystrophy of the bones and osteolysis of the hallux. At age 6, a spontaneous fracture of the right tibia was treated with surgery. Histopathology of a bone sample demonstrated bone remodelling, fibrous tissue, and large vascular lacunas within bone tissue, bordered by cells expressing the lymphaticmarker D2–40. At 8 years old, lymphedema of the right inferior leg had increased, leg lengths still differed, but other clinical signs were absent.
Url:
DOI: 10.1111/j.1525-1470.2011.01712.x
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<correspondenceTo>Address correspondence to Annabel Maruani, M.D., Ph.D., Department of Dermatology, Trousseau Hospital, CHRU Tours, Avenue de la République, 37044 Tours Cedex 9, France, or e‐mail: <email normalForm="annabel.maruani@univ-tours.fr">annabel.maruani@univ-tours.fr</email>
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<contentMeta><countGroup><count type="figureTotal" number="4"></count>
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<titleGroup><title type="main">Treatment of EARLY‐ONSET Gorham Syndrome with 8‐Year Follow‐Up</title>
<title type="short">Brief Report</title>
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<degrees>M.D., Ph.D.</degrees>
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<creator creatorRole="author" xml:id="cr2" affiliationRef="#a1 #a5"><personName><givenNames>Sylvie</givenNames>
<familyName>Thimon</familyName>
<degrees>M.D.</degrees>
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<creator creatorRole="author" xml:id="cr3" affiliationRef="#a2 #a5"><personName><givenNames>Benoît</givenNames>
<familyName>de Courtivron</familyName>
<degrees>M.D.</degrees>
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<creator creatorRole="author" xml:id="cr4" affiliationRef="#a3 #a5"><personName><givenNames>Gonzague</givenNames>
<familyName>de Pinieux</familyName>
<degrees>M.D.</degrees>
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<creator creatorRole="author" xml:id="cr5" affiliationRef="#a4 #a5"><personName><givenNames>Françoise</givenNames>
<familyName>Baulieu</familyName>
<degrees>M.D.</degrees>
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<creator creatorRole="author" xml:id="cr6" affiliationRef="#a1 #a5 #a6"><personName><givenNames>Laurent</givenNames>
<familyName>Machet</familyName>
<degrees>M.D., Ph.D.</degrees>
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<creator creatorRole="author" xml:id="cr7" affiliationRef="#a1 #a5"><personName><givenNames>Gérard</givenNames>
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<affiliationGroup><affiliation xml:id="a1"><unparsedAffiliation>Departments of Dermatology</unparsedAffiliation>
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<affiliation xml:id="a2"><unparsedAffiliation>Pediatric Orthopaedic Surgery</unparsedAffiliation>
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<affiliation xml:id="a3"><unparsedAffiliation>Pathology, and</unparsedAffiliation>
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<affiliation xml:id="a4"><unparsedAffiliation>Nuclear Medicine, University François Rabelais Tours</unparsedAffiliation>
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<affiliation xml:id="a5"><unparsedAffiliation>CHRU Tours</unparsedAffiliation>
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<abstractGroup><abstract type="main" xml:lang="en"><p><b>Abstract: </b>
A case of Gorham disease with several years of follow‐up is reported. At birth he had a mass in the thigh which was had pathology demonstrating a lymphangioma. By age 3 years, he had lymphedema of the ipsilateral foot and discrepant leg lengths. Radiography revealed heterogenous dystrophy of the bones and osteolysis of the hallux. At age 6, a spontaneous fracture of the right tibia was treated with surgery. Histopathology of a bone sample demonstrated bone remodelling, fibrous tissue, and large vascular lacunas within bone tissue, bordered by cells expressing the lymphaticmarker D2–40. At 8 years old, lymphedema of the right inferior leg had increased, leg lengths still differed, but other clinical signs were absent.</p>
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<titleInfo type="abbreviated" lang="en"><title>Brief Report</title>
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<affiliation>CHRU Tours</affiliation>
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<name type="personal"><namePart type="given">Laurent</namePart>
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<affiliation>CHRU Tours</affiliation>
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<dateIssued encoding="w3cdtf">2013-05</dateIssued>
<copyrightDate encoding="w3cdtf">2013</copyrightDate>
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<abstract>Abstract: A case of Gorham disease with several years of follow‐up is reported. At birth he had a mass in the thigh which was had pathology demonstrating a lymphangioma. By age 3 years, he had lymphedema of the ipsilateral foot and discrepant leg lengths. Radiography revealed heterogenous dystrophy of the bones and osteolysis of the hallux. At age 6, a spontaneous fracture of the right tibia was treated with surgery. Histopathology of a bone sample demonstrated bone remodelling, fibrous tissue, and large vascular lacunas within bone tissue, bordered by cells expressing the lymphaticmarker D2–40. At 8 years old, lymphedema of the right inferior leg had increased, leg lengths still differed, but other clinical signs were absent.</abstract>
<relatedItem type="host"><titleInfo><title>Pediatric Dermatology</title>
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<topic>BRIEF REPORT</topic>
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<identifier type="ISSN">0736-8046</identifier>
<identifier type="eISSN">1525-1470</identifier>
<identifier type="DOI">10.1111/(ISSN)1525-1470</identifier>
<identifier type="PublisherID">PDE</identifier>
<part><date>2013</date>
<detail type="volume"><caption>vol.</caption>
<number>30</number>
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<detail type="issue"><caption>no.</caption>
<number>3</number>
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<identifier type="DOI">10.1111/j.1525-1470.2011.01712.x</identifier>
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