Sirolimus and Autophagy Inhibition in Lymphangioleiomyomatosis: Results of a Phase I Clinical Trial.
Identifieur interne : 000177 ( PubMed/Corpus ); précédent : 000176; suivant : 000178Sirolimus and Autophagy Inhibition in Lymphangioleiomyomatosis: Results of a Phase I Clinical Trial.
Auteurs : Souheil El-Chemaly ; Angelo Taveira-Dasilva ; Hilary J. Goldberg ; Elizabeth Peters ; Mary Haughey ; Don Bienfang ; Amanda M. Jones ; Patricia Julien-Williams ; Ye Cui ; Julian A. Villalba ; Shefali Bagwe ; Rie Maurer ; Ivan O. Rosas ; Joel Moss ; Elizabeth P. HenskeSource :
- Chest [ 1931-3543 ] ; 2017.
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
Links to Exploration step
pubmed:28192114Le document en format XML
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Electronic address: sel-chemaly@partners.org.</nlm:affiliation></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><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation></affiliation></author><author><name sortKey="Goldberg, Hilary J" sort="Goldberg, Hilary J" uniqKey="Goldberg H" first="Hilary J" last="Goldberg">Hilary J. 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Electronic address: sel-chemaly@partners.org.</nlm:affiliation></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><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation></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><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></affiliation></author><author><name sortKey="Peters, Elizabeth" sort="Peters, Elizabeth" uniqKey="Peters E" first="Elizabeth" last="Peters">Elizabeth Peters</name><affiliation><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></affiliation></author><author><name sortKey="Haughey, Mary" sort="Haughey, Mary" uniqKey="Haughey M" first="Mary" last="Haughey">Mary Haughey</name><affiliation><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation></affiliation></author><author><name sortKey="Bienfang, Don" sort="Bienfang, Don" uniqKey="Bienfang D" first="Don" last="Bienfang">Don Bienfang</name><affiliation><nlm:affiliation>Department of Neurology, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></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><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation></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><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation></affiliation></author><author><name sortKey="Cui, Ye" sort="Cui, Ye" uniqKey="Cui Y" first="Ye" last="Cui">Ye Cui</name><affiliation><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></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><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></affiliation></author><author><name sortKey="Bagwe, Shefali" sort="Bagwe, Shefali" uniqKey="Bagwe S" first="Shefali" last="Bagwe">Shefali Bagwe</name><affiliation><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></affiliation></author><author><name sortKey="Maurer, Rie" sort="Maurer, Rie" uniqKey="Maurer R" first="Rie" last="Maurer">Rie Maurer</name><affiliation><nlm:affiliation>Harvard Clinical and Translational Science Center, Boston, MA.</nlm:affiliation></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><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></affiliation></author><author><name sortKey="Moss, Joel" sort="Moss, Joel" uniqKey="Moss J" first="Joel" last="Moss">Joel Moss</name><affiliation><nlm:affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</nlm:affiliation></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><nlm:affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</nlm:affiliation></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="MESH" type="chemical" qualifier="administration & dosage" xml:lang="en"><term>Enzyme Inhibitors</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="physiopathology" xml:lang="en"><term>Lymphangioleiomyomatosis</term></keywords><keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>Immunosuppressive Agents</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></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><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">28192114</PMID><DateCompleted><Year>2017</Year><Month>08</Month><Day>21</Day></DateCompleted><DateRevised><Year>2018</Year><Month>11</Month><Day>13</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1931-3543</ISSN><JournalIssue CitedMedium="Internet"><Volume>151</Volume><Issue>6</Issue><PubDate><Year>2017</Year><Month>06</Month></PubDate></JournalIssue><Title>Chest</Title><ISOAbbreviation>Chest</ISOAbbreviation></Journal><ArticleTitle>Sirolimus and Autophagy Inhibition in Lymphangioleiomyomatosis: Results of a Phase I Clinical Trial.</ArticleTitle><Pagination><MedlinePgn>1302-1310</MedlinePgn></Pagination><ELocationID EIdType="pii" ValidYN="Y">S0012-3692(17)30174-5</ELocationID><ELocationID EIdType="doi" ValidYN="Y">10.1016/j.chest.2017.01.033</ELocationID><Abstract><AbstractText Label="BACKGROUND">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.</AbstractText><AbstractText Label="METHODS">This 48-week, two-center phase I trial evaluated the safety of escalating oral hydroxychloroquine doses (100-200 mg) given twice a day in combination with sirolimus to eligible patients ≥ 18 years old with LAM. Subjects received combination therapy for 24 weeks followed by an observation phase without taking study drugs for an additional 24 weeks.</AbstractText><AbstractText Label="RESULTS">Fourteen patients provided written informed consent. Thirteen were treated in cohorts of three patients each with escalating hydroxychloroquine doses (200 and 400 mg) and an extension phase at the 400-mg dose. The most common adverse events were mucositis, headache, and diarrhea. No drug-related serious adverse events were reported. Secondary end points showed improvement in lung function at 24 weeks, with a decrease in lung function at the 48-week time point. When the higher dose of hydroxychloroquine was analyzed separately, FEV<sub>1</sub> and FVC remained stable at 48 weeks, but the 6-min walk distance showed a decrease toward baseline.</AbstractText><AbstractText Label="CONCLUSIONS">The combination of sirolimus and hydroxychloroquine is well tolerated, with no dose-limiting adverse events observed at 200 mg twice a day. Potential effects on lung function should be explored in larger trials.</AbstractText><AbstractText Label="TRIAL REGISTRY">ClinicalTrials.gov; No.: NCT01687179; URL: www.clinicaltrials.gov.</AbstractText><CopyrightInformation>Copyright © 2017 American College of Chest Physicians. All rights reserved.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>El-Chemaly</LastName><ForeName>Souheil</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA. Electronic address: sel-chemaly@partners.org.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Taveira-Dasilva</LastName><ForeName>Angelo</ForeName><Initials>A</Initials><AffiliationInfo><Affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Goldberg</LastName><ForeName>Hilary J</ForeName><Initials>HJ</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Peters</LastName><ForeName>Elizabeth</ForeName><Initials>E</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Haughey</LastName><ForeName>Mary</ForeName><Initials>M</Initials><AffiliationInfo><Affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Bienfang</LastName><ForeName>Don</ForeName><Initials>D</Initials><AffiliationInfo><Affiliation>Department of Neurology, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Jones</LastName><ForeName>Amanda M</ForeName><Initials>AM</Initials><AffiliationInfo><Affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Julien-Williams</LastName><ForeName>Patricia</ForeName><Initials>P</Initials><AffiliationInfo><Affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Cui</LastName><ForeName>Ye</ForeName><Initials>Y</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Villalba</LastName><ForeName>Julian A</ForeName><Initials>JA</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Bagwe</LastName><ForeName>Shefali</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Maurer</LastName><ForeName>Rie</ForeName><Initials>R</Initials><AffiliationInfo><Affiliation>Harvard Clinical and Translational Science Center, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Rosas</LastName><ForeName>Ivan O</ForeName><Initials>IO</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Moss</LastName><ForeName>Joel</ForeName><Initials>J</Initials><AffiliationInfo><Affiliation>Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, MD.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Henske</LastName><ForeName>Elizabeth P</ForeName><Initials>EP</Initials><AffiliationInfo><Affiliation>Division of Pulmonary and Critical Care Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><DataBankList CompleteYN="Y"><DataBank><DataBankName>ClinicalTrials.gov</DataBankName><AccessionNumberList><AccessionNumber>NCT01687179</AccessionNumber></AccessionNumberList></DataBank></DataBankList><PublicationTypeList><PublicationType UI="D017426">Clinical Trial, Phase I</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D052060">Research Support, N.I.H., Intramural</PublicationType><PublicationType UI="D013486">Research Support, U.S. Gov't, Non-P.H.S.</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2017</Year><Month>02</Month><Day>10</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Chest</MedlineTA><NlmUniqueID>0231335</NlmUniqueID><ISSNLinking>0012-3692</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D004791">Enzyme Inhibitors</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D007166">Immunosuppressive Agents</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="C579459">VEGFD protein, human</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D042643">Vascular Endothelial Growth Factor D</NameOfSubstance></Chemical><Chemical><RegistryNumber>4QWG6N8QKH</RegistryNumber><NameOfSubstance UI="D006886">Hydroxychloroquine</NameOfSubstance></Chemical><Chemical><RegistryNumber>W36ZG6FT64</RegistryNumber><NameOfSubstance UI="D020123">Sirolimus</NameOfSubstance></Chemical></ChemicalList><CitationSubset>AIM</CitationSubset><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName UI="D000328" MajorTopicYN="N">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D000368" MajorTopicYN="N">Aged</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D001343" MajorTopicYN="N">Autophagy</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D003967" MajorTopicYN="N">Diarrhea</DescriptorName><QualifierName UI="Q000139" MajorTopicYN="N">chemically induced</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D004791" MajorTopicYN="N">Enzyme Inhibitors</DescriptorName><QualifierName UI="Q000008" MajorTopicYN="Y">administration & dosage</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D004797" MajorTopicYN="N">Enzyme-Linked Immunosorbent Assay</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005541" MajorTopicYN="N">Forced Expiratory Volume</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006261" MajorTopicYN="N">Headache</DescriptorName><QualifierName UI="Q000139" MajorTopicYN="N">chemically induced</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006886" MajorTopicYN="N">Hydroxychloroquine</DescriptorName><QualifierName UI="Q000008" MajorTopicYN="Y">administration & dosage</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D007166" MajorTopicYN="N">Immunosuppressive Agents</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="Y">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D018192" MajorTopicYN="N">Lymphangioleiomyomatosis</DescriptorName><QualifierName UI="Q000097" 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