Sirolimus and Autophagy Inhibition in Lymphangioleiomyomatosis: Results of a Phase I Clinical Trial.
Identifieur interne : 000C95 ( Main/Exploration ); précédent : 000C94; suivant : 000C96Sirolimus and Autophagy Inhibition in Lymphangioleiomyomatosis: Results of a Phase I Clinical Trial.
Auteurs : Souheil El-Chemaly [États-Unis] ; Angelo Taveira-Dasilva [États-Unis] ; Hilary J. Goldberg [États-Unis] ; Elizabeth Peters [États-Unis] ; Mary Haughey [États-Unis] ; Don Bienfang [États-Unis] ; Amanda M. Jones [États-Unis] ; Patricia Julien-Williams [États-Unis] ; Ye Cui [États-Unis] ; Julian A. Villalba [États-Unis] ; Shefali Bagwe [États-Unis] ; Rie Maurer [États-Unis] ; Ivan O. Rosas [États-Unis] ; Joel Moss [États-Unis] ; Elizabeth P. Henske [États-Unis]Source :
- Chest [ 1931-3543 ] ; 2017.
Descripteurs français
- KwdFr :
- Adulte, Adulte d'âge moyen, Antienzymes (administration et posologie), Autophagie, Capacité vitale, Céphalée (), Diarrhée (), Facteur de croissance endothéliale vasculaire de type D (sang), Femelle, Humains, Hydroxychloroquine (administration et posologie), Immunosuppresseurs (usage thérapeutique), Inflammation muqueuse (), Lymphangioléiomyomatose (physiopathologie), Lymphangioléiomyomatose (sang), Lymphangioléiomyomatose (traitement médicamenteux), Résultat thérapeutique, Sirolimus (usage thérapeutique), Sujet âgé, Test ELISA, Test de marche, Volume expiratoire maximal par seconde.
- MESH :
- administration et posologie : Antienzymes, Hydroxychloroquine.
- physiopathologie : Lymphangioléiomyomatose.
- sang : Facteur de croissance endothéliale vasculaire de type D, Lymphangioléiomyomatose.
- traitement médicamenteux : Lymphangioléiomyomatose.
- usage thérapeutique : Immunosuppresseurs, Sirolimus.
- Adulte, Adulte d'âge moyen, Autophagie, Capacité vitale, Céphalée, Diarrhée, Femelle, Humains, Inflammation muqueuse, Résultat thérapeutique, Sujet âgé, Test ELISA, Test de marche, Volume expiratoire maximal par seconde.
English descriptors
- KwdEn :
- Adult, Aged, Autophagy, Diarrhea (chemically induced), Enzyme Inhibitors (administration & dosage), Enzyme-Linked Immunosorbent Assay, Female, Forced Expiratory Volume, Headache (chemically induced), Humans, Hydroxychloroquine (administration & dosage), Immunosuppressive Agents (therapeutic use), Lymphangioleiomyomatosis (blood), Lymphangioleiomyomatosis (drug therapy), Lymphangioleiomyomatosis (physiopathology), Middle Aged, Mucositis (chemically induced), Sirolimus (therapeutic use), Treatment Outcome, Vascular Endothelial Growth Factor D (blood), Vital Capacity, Walk Test.
- MESH :
- chemical , administration & dosage : Enzyme Inhibitors, Hydroxychloroquine.
- blood : Lymphangioleiomyomatosis, Vascular Endothelial Growth Factor D.
- chemically induced : Diarrhea, Headache, Mucositis.
- drug therapy : Lymphangioleiomyomatosis.
- physiopathology : Lymphangioleiomyomatosis.
- chemical , therapeutic use : Immunosuppressive Agents, Sirolimus.
- Adult, Aged, Autophagy, Enzyme-Linked Immunosorbent Assay, Female, Forced Expiratory Volume, Humans, Middle Aged, Treatment Outcome, Vital Capacity, Walk Test.
Abstract
Animal and cellular studies support the importance of autophagy inhibition in lymphangioleiomyomatosis (LAM). In a cohort of subjects with LAM, we tested the hypothesis that treatment with sirolimus and hydroxychloroquine (an autophagy inhibitor) at two different dose levels is safe and well tolerated. Secondary end points included changes in lung function.
DOI: 10.1016/j.chest.2017.01.033
PubMed: 28192114
Affiliations:
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Le document en format XML
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Electronic address: sel-chemaly@partners.org.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Taveira Dasilva, Angelo" sort="Taveira Dasilva, Angelo" uniqKey="Taveira Dasilva A" first="Angelo" last="Taveira-Dasilva">Angelo Taveira-Dasilva</name><affiliation wicri:level="2"><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Maryland</region></placeName><wicri:cityArea>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda</wicri:cityArea></affiliation></author><author><name sortKey="Goldberg, Hilary J" sort="Goldberg, Hilary J" uniqKey="Goldberg H" first="Hilary J" last="Goldberg">Hilary J. Goldberg</name><affiliation wicri:level="2"><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Peters, Elizabeth" sort="Peters, Elizabeth" uniqKey="Peters E" first="Elizabeth" last="Peters">Elizabeth Peters</name><affiliation wicri:level="2"><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Haughey, Mary" sort="Haughey, Mary" uniqKey="Haughey M" first="Mary" last="Haughey">Mary Haughey</name><affiliation wicri:level="2"><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Maryland</region></placeName><wicri:cityArea>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda</wicri:cityArea></affiliation></author><author><name sortKey="Bienfang, Don" sort="Bienfang, Don" uniqKey="Bienfang D" first="Don" last="Bienfang">Don Bienfang</name><affiliation wicri:level="2"><nlm:affiliation>Department of Neurology, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Department of Neurology, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Jones, Amanda M" sort="Jones, Amanda M" uniqKey="Jones A" first="Amanda M" last="Jones">Amanda M. Jones</name><affiliation wicri:level="2"><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Maryland</region></placeName><wicri:cityArea>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda</wicri:cityArea></affiliation></author><author><name sortKey="Julien Williams, Patricia" sort="Julien Williams, Patricia" uniqKey="Julien Williams P" first="Patricia" last="Julien-Williams">Patricia Julien-Williams</name><affiliation wicri:level="2"><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Maryland</region></placeName><wicri:cityArea>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda</wicri:cityArea></affiliation></author><author><name sortKey="Cui, Ye" sort="Cui, Ye" uniqKey="Cui Y" first="Ye" last="Cui">Ye Cui</name><affiliation wicri:level="2"><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Villalba, Julian A" sort="Villalba, Julian A" uniqKey="Villalba J" first="Julian A" last="Villalba">Julian A. Villalba</name><affiliation wicri:level="2"><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Bagwe, Shefali" sort="Bagwe, Shefali" uniqKey="Bagwe S" first="Shefali" last="Bagwe">Shefali Bagwe</name><affiliation wicri:level="2"><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Maurer, Rie" sort="Maurer, Rie" uniqKey="Maurer R" first="Rie" last="Maurer">Rie Maurer</name><affiliation wicri:level="2"><nlm:affiliation>Harvard Clinical and Translational Science Center, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Harvard Clinical and Translational Science Center, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Rosas, Ivan O" sort="Rosas, Ivan O" uniqKey="Rosas I" first="Ivan O" last="Rosas">Ivan O. Rosas</name><affiliation wicri:level="2"><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author><author><name sortKey="Moss, Joel" sort="Moss, Joel" uniqKey="Moss J" first="Joel" last="Moss">Joel Moss</name><affiliation wicri:level="2"><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Maryland</region></placeName><wicri:cityArea>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda</wicri:cityArea></affiliation></author><author><name sortKey="Henske, Elizabeth P" sort="Henske, Elizabeth P" uniqKey="Henske E" first="Elizabeth P" last="Henske">Elizabeth P. Henske</name><affiliation wicri:level="2"><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Massachusetts</region></placeName><wicri:cityArea>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston</wicri:cityArea></affiliation></author></analytic><series><title level="j">Chest</title><idno type="eISSN">1931-3543</idno><imprint><date when="2017" type="published">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Aged</term><term>Autophagy</term><term>Diarrhea (chemically induced)</term><term>Enzyme Inhibitors (administration & dosage)</term><term>Enzyme-Linked Immunosorbent Assay</term><term>Female</term><term>Forced Expiratory Volume</term><term>Headache (chemically induced)</term><term>Humans</term><term>Hydroxychloroquine (administration & dosage)</term><term>Immunosuppressive Agents (therapeutic use)</term><term>Lymphangioleiomyomatosis (blood)</term><term>Lymphangioleiomyomatosis (drug therapy)</term><term>Lymphangioleiomyomatosis (physiopathology)</term><term>Middle Aged</term><term>Mucositis (chemically induced)</term><term>Sirolimus (therapeutic use)</term><term>Treatment Outcome</term><term>Vascular Endothelial Growth Factor D (blood)</term><term>Vital Capacity</term><term>Walk Test</term></keywords><keywords scheme="KwdFr" xml:lang="fr"><term>Adulte</term><term>Adulte d'âge moyen</term><term>Antienzymes (administration et posologie)</term><term>Autophagie</term><term>Capacité vitale</term><term>Céphalée ()</term><term>Diarrhée ()</term><term>Facteur de croissance endothéliale vasculaire de type D (sang)</term><term>Femelle</term><term>Humains</term><term>Hydroxychloroquine (administration et posologie)</term><term>Immunosuppresseurs (usage thérapeutique)</term><term>Inflammation muqueuse ()</term><term>Lymphangioléiomyomatose (physiopathologie)</term><term>Lymphangioléiomyomatose (sang)</term><term>Lymphangioléiomyomatose (traitement médicamenteux)</term><term>Résultat thérapeutique</term><term>Sirolimus (usage thérapeutique)</term><term>Sujet âgé</term><term>Test ELISA</term><term>Test de marche</term><term>Volume expiratoire maximal par seconde</term></keywords><keywords scheme="MESH" type="chemical" qualifier="administration & dosage" xml:lang="en"><term>Enzyme Inhibitors</term><term>Hydroxychloroquine</term></keywords><keywords scheme="MESH" qualifier="administration et posologie" xml:lang="fr"><term>Antienzymes</term><term>Hydroxychloroquine</term></keywords><keywords scheme="MESH" qualifier="blood" xml:lang="en"><term>Lymphangioleiomyomatosis</term><term>Vascular Endothelial Growth Factor D</term></keywords><keywords scheme="MESH" qualifier="chemically induced" xml:lang="en"><term>Diarrhea</term><term>Headache</term><term>Mucositis</term></keywords><keywords scheme="MESH" qualifier="drug therapy" xml:lang="en"><term>Lymphangioleiomyomatosis</term></keywords><keywords scheme="MESH" qualifier="physiopathologie" xml:lang="fr"><term>Lymphangioléiomyomatose</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Lymphangioleiomyomatosis</term></keywords><keywords scheme="MESH" qualifier="sang" xml:lang="fr"><term>Facteur de croissance endothéliale vasculaire de type D</term><term>Lymphangioléiomyomatose</term></keywords><keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>Immunosuppressive Agents</term><term>Sirolimus</term></keywords><keywords scheme="MESH" qualifier="traitement médicamenteux" xml:lang="fr"><term>Lymphangioléiomyomatose</term></keywords><keywords scheme="MESH" qualifier="usage thérapeutique" xml:lang="fr"><term>Immunosuppresseurs</term><term>Sirolimus</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Aged</term><term>Autophagy</term><term>Enzyme-Linked Immunosorbent Assay</term><term>Female</term><term>Forced Expiratory Volume</term><term>Humans</term><term>Middle Aged</term><term>Treatment Outcome</term><term>Vital Capacity</term><term>Walk Test</term></keywords><keywords scheme="MESH" xml:lang="fr"><term>Adulte</term><term>Adulte d'âge moyen</term><term>Autophagie</term><term>Capacité vitale</term><term>Céphalée</term><term>Diarrhée</term><term>Femelle</term><term>Humains</term><term>Inflammation muqueuse</term><term>Résultat thérapeutique</term><term>Sujet âgé</term><term>Test ELISA</term><term>Test de marche</term><term>Volume expiratoire maximal par seconde</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Animal and cellular studies support the importance of autophagy inhibition in lymphangioleiomyomatosis (LAM). In a cohort of subjects with LAM, we tested the hypothesis that treatment with sirolimus and hydroxychloroquine (an autophagy inhibitor) at two different dose levels is safe and well tolerated. Secondary end points included changes in lung function.</div></front></TEI><affiliations><list><country><li>États-Unis</li></country><region><li>Maryland</li><li>Massachusetts</li></region></list><tree><country name="États-Unis"><region name="Massachusetts"><name sortKey="El Chemaly, Souheil" sort="El Chemaly, Souheil" uniqKey="El Chemaly S" first="Souheil" last="El-Chemaly">Souheil El-Chemaly</name></region><name sortKey="Bagwe, Shefali" sort="Bagwe, Shefali" uniqKey="Bagwe S" first="Shefali" last="Bagwe">Shefali Bagwe</name><name sortKey="Bienfang, Don" sort="Bienfang, Don" uniqKey="Bienfang D" first="Don" last="Bienfang">Don Bienfang</name><name sortKey="Cui, Ye" sort="Cui, Ye" uniqKey="Cui Y" first="Ye" last="Cui">Ye Cui</name><name sortKey="Goldberg, Hilary J" sort="Goldberg, Hilary J" uniqKey="Goldberg H" first="Hilary J" last="Goldberg">Hilary J. Goldberg</name><name sortKey="Haughey, Mary" sort="Haughey, Mary" uniqKey="Haughey M" first="Mary" last="Haughey">Mary Haughey</name><name sortKey="Henske, Elizabeth P" sort="Henske, Elizabeth P" uniqKey="Henske E" first="Elizabeth P" last="Henske">Elizabeth P. Henske</name><name sortKey="Jones, Amanda M" sort="Jones, Amanda M" uniqKey="Jones A" first="Amanda M" last="Jones">Amanda M. Jones</name><name sortKey="Julien Williams, Patricia" sort="Julien Williams, Patricia" uniqKey="Julien Williams P" first="Patricia" last="Julien-Williams">Patricia Julien-Williams</name><name sortKey="Maurer, Rie" sort="Maurer, Rie" uniqKey="Maurer R" first="Rie" last="Maurer">Rie Maurer</name><name sortKey="Moss, Joel" sort="Moss, Joel" uniqKey="Moss J" first="Joel" last="Moss">Joel Moss</name><name sortKey="Peters, Elizabeth" sort="Peters, Elizabeth" uniqKey="Peters E" first="Elizabeth" last="Peters">Elizabeth Peters</name><name sortKey="Rosas, Ivan O" sort="Rosas, Ivan O" uniqKey="Rosas I" first="Ivan O" last="Rosas">Ivan O. Rosas</name><name sortKey="Taveira Dasilva, Angelo" sort="Taveira Dasilva, Angelo" uniqKey="Taveira Dasilva A" first="Angelo" last="Taveira-Dasilva">Angelo Taveira-Dasilva</name><name sortKey="Villalba, Julian A" sort="Villalba, Julian A" uniqKey="Villalba J" first="Julian A" last="Villalba">Julian A. Villalba</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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