Use of somatostatin analogues to treat chylothorax in a child with Generalised Lymphatic Dysplasia.
Identifieur interne : 001D58 ( PubMed/Checkpoint ); précédent : 001D57; suivant : 001D59Use of somatostatin analogues to treat chylothorax in a child with Generalised Lymphatic Dysplasia.
Auteurs : Malcolm Brodlie [Royaume-Uni] ; Sara Abdelgalil [Royaume-Uni] ; Sahar Mansour [Royaume-Uni] ; David A. Spencer [Royaume-Uni]Source :
- Respiratory medicine case reports [ 2213-0071 ] ; 2012.
Abstract
Generalised Lymphatic Dysplasia is a rare condition that may be associated with significant chylothoraces. The management of such effusions is often challenging. We present the case of a 15-year-old girl with bilateral chylothoraces and lymphoedema of her limbs. A clinical diagnosis of Generalised Lymphatic Dysplasia was made and long-term treatment with somatostatin analogues (somatostatin initially followed by monthly octreotide) was initiated. Over 12 months there was symptomatic benefit with some objective improvement in lung function and no adverse effects. After a year of treatment there was some reaccumulation of fluid, however this did not require any intervention. This is the first paediatric report of the use of somatostatin analogues to manage chylothorax in Generalised Lymphatic Dysplasia and we conclude that they represent a potentially useful treatment modality. Experience is only anecdotal however and further studies are required to establish an evidence base with regard to efficacy and safety.
DOI: 10.1016/j.rmedc.2011.07.005
PubMed: 26056769
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Spencer</name><affiliation wicri:level="1"><nlm:affiliation>Department of Respiratory Paediatrics, Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne NE1 4LP, United Kingdom.</nlm:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Respiratory Paediatrics, Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne NE1 4LP</wicri:regionArea><wicri:noRegion>Newcastle upon Tyne NE1 4LP</wicri:noRegion></affiliation></author></analytic><series><title level="j">Respiratory medicine case reports</title><idno type="ISSN">2213-0071</idno><imprint><date when="2012" type="published">2012</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Generalised Lymphatic Dysplasia is a rare condition that may be associated with significant chylothoraces. The management of such effusions is often challenging. We present the case of a 15-year-old girl with bilateral chylothoraces and lymphoedema of her limbs. A clinical diagnosis of Generalised Lymphatic Dysplasia was made and long-term treatment with somatostatin analogues (somatostatin initially followed by monthly octreotide) was initiated. Over 12 months there was symptomatic benefit with some objective improvement in lung function and no adverse effects. After a year of treatment there was some reaccumulation of fluid, however this did not require any intervention. This is the first paediatric report of the use of somatostatin analogues to manage chylothorax in Generalised Lymphatic Dysplasia and we conclude that they represent a potentially useful treatment modality. Experience is only anecdotal however and further studies are required to establish an evidence base with regard to efficacy and safety.</div></front></TEI><pubmed><MedlineCitation Status="PubMed-not-MEDLINE" Owner="NLM"><PMID Version="1">26056769</PMID><DateCreated><Year>2015</Year><Month>06</Month><Day>10</Day></DateCreated><DateCompleted><Year>2015</Year><Month>06</Month><Day>10</Day></DateCompleted><DateRevised><Year>2017</Year><Month>09</Month><Day>02</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Print">2213-0071</ISSN><JournalIssue CitedMedium="Print"><Volume>5</Volume><PubDate><Year>2012</Year></PubDate></JournalIssue><Title>Respiratory medicine case reports</Title><ISOAbbreviation>Respir Med Case Rep</ISOAbbreviation></Journal><ArticleTitle>Use of somatostatin analogues to treat chylothorax in a child with Generalised Lymphatic Dysplasia.</ArticleTitle><Pagination><MedlinePgn>76-7</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1016/j.rmedc.2011.07.005</ELocationID><Abstract><AbstractText>Generalised Lymphatic Dysplasia is a rare condition that may be associated with significant chylothoraces. The management of such effusions is often challenging. We present the case of a 15-year-old girl with bilateral chylothoraces and lymphoedema of her limbs. A clinical diagnosis of Generalised Lymphatic Dysplasia was made and long-term treatment with somatostatin analogues (somatostatin initially followed by monthly octreotide) was initiated. Over 12 months there was symptomatic benefit with some objective improvement in lung function and no adverse effects. After a year of treatment there was some reaccumulation of fluid, however this did not require any intervention. This is the first paediatric report of the use of somatostatin analogues to manage chylothorax in Generalised Lymphatic Dysplasia and we conclude that they represent a potentially useful treatment modality. Experience is only anecdotal however and further studies are required to establish an evidence base with regard to efficacy and safety.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Brodlie</LastName><ForeName>Malcolm</ForeName><Initials>M</Initials><AffiliationInfo><Affiliation>Department of Respiratory Paediatrics, Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne NE1 4LP, United Kingdom ; Institute of Cellular Medicine, Newcastle University, Newcastle upon Tyne NE2 4HH, United Kingdom.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Abdelgalil</LastName><ForeName>Sara</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>Department of Respiratory Paediatrics, Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne NE1 4LP, United Kingdom.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Mansour</LastName><ForeName>Sahar</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>South West Thames Regional Genetics Unit, St George's, University of London, Cranmer Terrace, London SW17 0RE, United Kingdom.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Spencer</LastName><ForeName>David A</ForeName><Initials>DA</Initials><AffiliationInfo><Affiliation>Department of Respiratory Paediatrics, Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne NE1 4LP, United Kingdom.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><GrantList CompleteYN="Y"><Grant><GrantID>MRC_G0800419</GrantID><Agency>Medical Research Council</Agency><Country>United Kingdom</Country></Grant></GrantList><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2011</Year><Month>10</Month><Day>01</Day></ArticleDate></Article><MedlineJournalInfo><Country>England</Country><MedlineTA>Respir Med Case Rep</MedlineTA><NlmUniqueID>101604463</NlmUniqueID><ISSNLinking>2213-0071</ISSNLinking></MedlineJournalInfo><CommentsCorrectionsList><CommentsCorrections RefType="Cites"><RefSource>Pediatr Pulmonol. 2010 Jun;45(6):628; author reply 629-30</RefSource><PMID Version="1">20503291</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Ann Thorac Surg. 1998 Jul;66(1):253-4</RefSource><PMID Version="1">9692478</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Hum Genet. 2010 Feb;127(2):231-41</RefSource><PMID Version="1">19911200</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Ann Intern Med. 2004 Aug 3;141(3):246-7</RefSource><PMID Version="1">15289232</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>J Paediatr Child Health. 2004 Sep-Oct;40(9-10):585-8</RefSource><PMID Version="1">15367160</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Intensive Care Med. 2006 May;32(5):650-7</RefSource><PMID Version="1">16532329</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Clin Genet. 2010 May;77(5):438-52</RefSource><PMID Version="1">20447153</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Br J Dermatol. 2007 Jun;156(6):1230-4</RefSource><PMID Version="1">17459037</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Chest. 2008 Aug;134(2):375-81</RefSource><PMID Version="1">18403655</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Gastroenterology. 1999 Apr;116(4):1017-8</RefSource><PMID Version="1">10092335</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Pediatrics. 1985 Aug;76(2):206-18</RefSource><PMID Version="1">4022694</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Clin Respir J. 2007 Dec;1(2):120-1</RefSource><PMID Version="1">20298292</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Thorax. 2005 Mar;60(3):254-5</RefSource><PMID Version="1">15741446</PMID></CommentsCorrections></CommentsCorrectionsList><OtherID Source="NLM">PMC3920370</OtherID><KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">Children</Keyword><Keyword MajorTopicYN="N">Chylothorax</Keyword><Keyword MajorTopicYN="N">Generalised Lymphatic Dysplasia</Keyword><Keyword MajorTopicYN="N">Octreotide</Keyword><Keyword MajorTopicYN="N">Paediatrics</Keyword><Keyword MajorTopicYN="N">Somatostatin</Keyword></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2011</Year><Month>06</Month><Day>21</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2011</Year><Month>07</Month><Day>25</Day></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2015</Year><Month>6</Month><Day>10</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2012</Year><Month>1</Month><Day>1</Day><Hour>0</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2012</Year><Month>1</Month><Day>1</Day><Hour>0</Hour><Minute>1</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">26056769</ArticleId><ArticleId IdType="doi">10.1016/j.rmedc.2011.07.005</ArticleId><ArticleId IdType="pii">S1755-0017(11)00045-5</ArticleId><ArticleId IdType="pmc">PMC3920370</ArticleId></ArticleIdList></PubmedData></pubmed><affiliations><list><country><li>Royaume-Uni</li></country><region><li>Angleterre</li><li>Grand Londres</li></region><settlement><li>Londres</li></settlement><orgName><li>Université de Londres</li></orgName></list><tree><country name="Royaume-Uni"><noRegion><name sortKey="Brodlie, Malcolm" sort="Brodlie, Malcolm" uniqKey="Brodlie M" first="Malcolm" last="Brodlie">Malcolm Brodlie</name></noRegion><name sortKey="Abdelgalil, Sara" sort="Abdelgalil, Sara" uniqKey="Abdelgalil S" first="Sara" last="Abdelgalil">Sara Abdelgalil</name><name sortKey="Mansour, Sahar" sort="Mansour, Sahar" uniqKey="Mansour S" first="Sahar" last="Mansour">Sahar Mansour</name><name sortKey="Spencer, David A" sort="Spencer, David A" uniqKey="Spencer D" first="David A" last="Spencer">David A. 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