Use of somatostatin analogues to treat chylothorax in a child with Generalised Lymphatic Dysplasia
Identifieur interne : 004A13 ( Main/Exploration ); précédent : 004A12; suivant : 004A14Use 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 ] ; 2011.
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.
Url:
DOI: 10.1016/j.rmedc.2011.07.005
PubMed: 26056769
PubMed Central: 3920370
Affiliations:
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Spencer</name><affiliation wicri:level="1"><nlm:aff id="aff1">Department of Respiratory Paediatrics, Great North Children’s Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne NE1 4LP, United Kingdom</nlm:aff><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="eISSN">2213-0071</idno><imprint><date when="2011">2011</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>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.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Smeltzer, D M" uniqKey="Smeltzer D">D.M. Smeltzer</name></author><author><name sortKey="Stickler, G B" uniqKey="Stickler G">G.B. Stickler</name></author><author><name sortKey="Schirger, A" uniqKey="Schirger A">A. Schirger</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Connell, F" uniqKey="Connell F">F. Connell</name></author><author><name sortKey="Brice, G" uniqKey="Brice G">G. Brice</name></author><author><name sortKey="Jeffery, S" uniqKey="Jeffery S">S. Jeffery</name></author><author><name sortKey="Keeley, V" uniqKey="Keeley V">V. Keeley</name></author><author><name sortKey="Mortimer, P" uniqKey="Mortimer P">P. Mortimer</name></author><author><name sortKey="Mansour, S" uniqKey="Mansour S">S. 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Unsur</name></author></analytic></biblStruct></listBibl></div1></back></TEI><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="Brodlie, Malcolm" sort="Brodlie, Malcolm" uniqKey="Brodlie M" first="Malcolm" last="Brodlie">Malcolm Brodlie</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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