The efficacy and safety of pharmacological treatments for lymphangioleiomyomatosis
Identifieur interne : 000025 ( Main/Exploration ); précédent : 000024; suivant : 000026The efficacy and safety of pharmacological treatments for lymphangioleiomyomatosis
Auteurs : Qi Wang [République populaire de Chine] ; Mengqi Luo [République populaire de Chine] ; Bo Xiang [République populaire de Chine] ; Siyuan Chen [République populaire de Chine] ; Yi Ji [République populaire de Chine]Source :
- Respiratory Research [ 1465-9921 ] ; 2020.
Abstract
Lymphangioleiomyomatosis (LAM) is a rare, low-grade multisystem neoplastic disease
PubMed (Medline), EMBASE, Cochrane Library, Web of Science and EBSCO Host were searched (until March 31, 2019) for eligible prospective studies regarding LAM patients treated with pharmacological treatments. Random effect models were used for quantitative analysis.
Fourteen prospective studies regarding five pharmacological treatments (including sirolimus, everolimus, doxycycline, triptorelin, and a combination therapy of sirolimus and hydroxychloroquine) were enrolled in our systematic review, and ten of them were used for the meta-analysis. Seven prospective studies reported that sirolimus was effective at improving or stabilizing lung function and alleviating renal angiomyolipoma (AML) in LAM patients. Subsequent quantitative analyses showed that during sirolimus treatment, the pooled values of lung function and 6-min walk distance (6MWD) were not significantly changed (
Overall, sirolimus and everolimus were recommended for the treatment of LAM because they could stabilize lung function and alleviate renal AML. Doxycycline and triptorelin were not recommended for the treatment of LAM because no beneficial outcomes were consistently observed. The efficacy and safety of combination therapy remain to be further explored.
Url:
DOI: 10.1186/s12931-020-1316-3
PubMed: 32059669
PubMed Central: 7023761
Affiliations:
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Oncology, Department of Pediatric Surgery,</institution><institution>West China Hospital of Sichuan University,</institution></institution-wrap>#37# Guo-Xue-Xiang, Chengdu, 610041 China</nlm:aff><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>#37# Guo-Xue-Xiang, Chengdu</wicri:regionArea><wicri:noRegion>Chengdu</wicri:noRegion></affiliation></author><author><name sortKey="Luo, Mengqi" sort="Luo, Mengqi" uniqKey="Luo M" first="Mengqi" last="Luo">Mengqi Luo</name><affiliation wicri:level="1"><nlm:aff id="Aff2"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 0807 1581</institution-id><institution-id institution-id-type="GRID">grid.13291.38</institution-id><institution>State Key Laboratory of Oral Diseases, National Clinical Research Centre for Oral Diseases, West China Hospital of Stomatology,</institution><institution>Sichuan University,</institution></institution-wrap>Chengdu, China</nlm:aff><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Chengdu</wicri:regionArea></affiliation></author><author><name sortKey="Xiang, Bo" sort="Xiang, Bo" uniqKey="Xiang B" first="Bo" last="Xiang">Bo Xiang</name><affiliation wicri:level="1"><nlm:aff id="Aff1"><institution-wrap><institution-id institution-id-type="ISNI">0000 0004 1770 1022</institution-id><institution-id institution-id-type="GRID">grid.412901.f</institution-id><institution>Division of Oncology, Department of Pediatric Surgery,</institution><institution>West China Hospital of Sichuan University,</institution></institution-wrap>#37# Guo-Xue-Xiang, Chengdu, 610041 China</nlm:aff><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>#37# Guo-Xue-Xiang, Chengdu</wicri:regionArea><wicri:noRegion>Chengdu</wicri:noRegion></affiliation></author><author><name sortKey="Chen, Siyuan" sort="Chen, Siyuan" uniqKey="Chen S" first="Siyuan" last="Chen">Siyuan Chen</name><affiliation wicri:level="1"><nlm:aff id="Aff3"><institution-wrap><institution-id institution-id-type="ISNI">0000 0004 1770 1022</institution-id><institution-id institution-id-type="GRID">grid.412901.f</institution-id><institution>Pediatric Intensive Care Unit, Department of Critical Care Medicine,</institution><institution>West China Hospital of Sichuan University,</institution></institution-wrap>Chengdu, China</nlm:aff><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Chengdu</wicri:regionArea></affiliation></author><author><name sortKey="Ji, Yi" sort="Ji, Yi" uniqKey="Ji Y" first="Yi" last="Ji">Yi Ji</name><affiliation wicri:level="1"><nlm:aff id="Aff1"><institution-wrap><institution-id institution-id-type="ISNI">0000 0004 1770 1022</institution-id><institution-id institution-id-type="GRID">grid.412901.f</institution-id><institution>Division of Oncology, Department of Pediatric Surgery,</institution><institution>West China Hospital of Sichuan University,</institution></institution-wrap>#37# Guo-Xue-Xiang, Chengdu, 610041 China</nlm:aff><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>#37# Guo-Xue-Xiang, Chengdu</wicri:regionArea><wicri:noRegion>Chengdu</wicri:noRegion></affiliation></author></analytic><series><title level="j">Respiratory Research</title><idno type="ISSN">1465-9921</idno><idno type="eISSN">1465-993X</idno><imprint><date when="2020">2020</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec><title>Background</title><p id="Par1">Lymphangioleiomyomatosis (LAM) is a rare, low-grade multisystem neoplastic disease<bold>.</bold> Most LAM patients are at a high risk of losing lung function at an accelerated rate and developing progressive dyspnea. Recently, several studies have reported their experience with pharmacological treatments for LAM. Therefore, we conducted a systematic review and meta-analysis to assess the efficacy and safety of these therapies.</p></sec><sec><title>Methods</title><p id="Par2">PubMed (Medline), EMBASE, Cochrane Library, Web of Science and EBSCO Host were searched (until March 31, 2019) for eligible prospective studies regarding LAM patients treated with pharmacological treatments. Random effect models were used for quantitative analysis.</p></sec><sec><title>Results</title><p id="Par3">Fourteen prospective studies regarding five pharmacological treatments (including sirolimus, everolimus, doxycycline, triptorelin, and a combination therapy of sirolimus and hydroxychloroquine) were enrolled in our systematic review, and ten of them were used for the meta-analysis. Seven prospective studies reported that sirolimus was effective at improving or stabilizing lung function and alleviating renal angiomyolipoma (AML) in LAM patients. Subsequent quantitative analyses showed that during sirolimus treatment, the pooled values of lung function and 6-min walk distance (6MWD) were not significantly changed (<italic>P</italic> > 0.05), with the pooled response rate of AML being 0.62 (95% confidence intervals [CIs]: 0.43 to 0.82, <italic>I</italic><sup><italic>2</italic></sup> = 65%). Regarding everolimus, three prospective studies reported similar effects to those of sirolimus with regard to preserving lung function and reducing AMLs. The meta-analysis showed that the changes in lung function during everolimus treatment were not statistically significant (<italic>P</italic> > 0.05), while the pooled response rate of AML was 0.78 (95% CI: 0.68 to 0.88, <italic>I</italic><sup><italic>2</italic></sup> = 8%). Neither the qualitative nor the quantitative results confirmed the benefits of doxycycline or triptorelin treatment, and the effects of the combination therapy were unclear in LAM patients. Most of the adverse events during pharmacological treatments were low or moderate grade and tolerable.</p></sec><sec><title>Conclusions</title><p id="Par4">Overall, sirolimus and everolimus were recommended for the treatment of LAM because they could stabilize lung function and alleviate renal AML. Doxycycline and triptorelin were not recommended for the treatment of LAM because no beneficial outcomes were consistently observed. The efficacy and safety of combination therapy remain to be further explored.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Mccormack, Fx" uniqKey="Mccormack F">FX McCormack</name></author><author><name sortKey="Gupta, N" uniqKey="Gupta N">N Gupta</name></author><author><name sortKey="Finlay, Gr" uniqKey="Finlay G">GR Finlay</name></author><author><name sortKey="Young, Lr" uniqKey="Young L">LR Young</name></author><author><name sortKey="Taveira Dasilva, Am" uniqKey="Taveira Dasilva A">AM Taveira-DaSilva</name></author><author><name sortKey="Glasgow, Cg" uniqKey="Glasgow C">CG Glasgow</name></author><author><name sortKey="Steagall, Wk" uniqKey="Steagall W">WK Steagall</name></author><author><name sortKey="Johnson, Sr" uniqKey="Johnson S">SR Johnson</name></author><author><name sortKey="Sahn, Sa" uniqKey="Sahn S">SA Sahn</name></author><author><name sortKey="Ryu, Jh" uniqKey="Ryu J">JH 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Lewis</name></author><author><name sortKey="Kumaran, M" uniqKey="Kumaran M">M Kumaran</name></author><author><name sortKey="Cane, Jl" uniqKey="Cane J">JL Cane</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Young, L" uniqKey="Young L">L Young</name></author><author><name sortKey="Lee, Hs" uniqKey="Lee H">HS Lee</name></author><author><name sortKey="Inoue, Y" uniqKey="Inoue Y">Y Inoue</name></author><author><name sortKey="Moss, J" uniqKey="Moss J">J Moss</name></author><author><name sortKey="Singer, Lg" uniqKey="Singer L">LG Singer</name></author><author><name sortKey="Strange, C" uniqKey="Strange C">C Strange</name></author><author><name sortKey="Nakata, K" uniqKey="Nakata K">K Nakata</name></author><author><name sortKey="Barker, Af" uniqKey="Barker A">AF Barker</name></author><author><name sortKey="Chapman, Jt" uniqKey="Chapman J">JT Chapman</name></author><author><name sortKey="Brantly, Ml" uniqKey="Brantly M">ML Brantly</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Xu, Kf" uniqKey="Xu K">KF Xu</name></author><author><name sortKey="Tian, X" uniqKey="Tian X">X Tian</name></author><author><name sortKey="Yang, Y" uniqKey="Yang Y">Y Yang</name></author><author><name sortKey="Zhang, H" uniqKey="Zhang H">H Zhang</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Harari, S" uniqKey="Harari S">S Harari</name></author><author><name sortKey="Spagnolo, P" uniqKey="Spagnolo P">P Spagnolo</name></author><author><name sortKey="Cocconcelli, E" uniqKey="Cocconcelli E">E Cocconcelli</name></author><author><name sortKey="Luisi, F" uniqKey="Luisi F">F Luisi</name></author><author><name sortKey="Cottin, V" uniqKey="Cottin V">V Cottin</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Yao, J" uniqKey="Yao J">J Yao</name></author><author><name sortKey="Taveira Dasilva, Am" uniqKey="Taveira Dasilva A">AM Taveira-DaSilva</name></author><author><name sortKey="Jones, Am" uniqKey="Jones A">AM Jones</name></author><author><name sortKey="Julien Williams, P" uniqKey="Julien Williams P">P Julien-Williams</name></author><author><name sortKey="Stylianou, M" uniqKey="Stylianou M">M Stylianou</name></author><author><name sortKey="Moss, J" uniqKey="Moss J">J Moss</name></author></analytic></biblStruct></listBibl></div1></back></TEI><affiliations><list><country><li>République populaire de Chine</li></country></list><tree><country name="République populaire de Chine"><noRegion><name sortKey="Wang, Qi" sort="Wang, Qi" uniqKey="Wang Q" first="Qi" last="Wang">Qi Wang</name></noRegion><name sortKey="Chen, Siyuan" sort="Chen, Siyuan" uniqKey="Chen S" first="Siyuan" last="Chen">Siyuan Chen</name><name sortKey="Ji, Yi" sort="Ji, Yi" uniqKey="Ji Y" first="Yi" last="Ji">Yi Ji</name><name sortKey="Luo, Mengqi" sort="Luo, Mengqi" uniqKey="Luo M" first="Mengqi" last="Luo">Mengqi Luo</name><name sortKey="Xiang, Bo" sort="Xiang, Bo" uniqKey="Xiang B" first="Bo" last="Xiang">Bo Xiang</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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