The efficacy and safety of pharmacological treatments for lymphangioleiomyomatosis.
Identifieur interne : 000005 ( PubMed/Curation ); précédent : 000004; suivant : 000006The 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 [ 1465-993X ] ; 2020.
Abstract
Lymphangioleiomyomatosis (LAM) is a rare, low-grade multisystem neoplastic disease. 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.
DOI: 10.1186/s12931-020-1316-3
PubMed: 32059669
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Stomatology, Sichuan University, Chengdu, China.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>State Key Laboratory of Oral Diseases, National Clinical Research Centre for Oral Diseases, West China Hospital of Stomatology, Sichuan University, 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:affiliation>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041</wicri:regionArea></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:affiliation>Pediatric Intensive Care Unit, Department of Critical Care Medicine, West China Hospital of Sichuan University, Chengdu, China.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Pediatric Intensive Care Unit, Department of Critical Care Medicine, West China Hospital of Sichuan University, 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:affiliation>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China. jijiyuanyuan@163.com.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 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Chengdu, 610041, China.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041</wicri:regionArea></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:affiliation>State Key Laboratory of Oral Diseases, National Clinical Research Centre for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu, China.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>State Key Laboratory of Oral Diseases, National Clinical Research Centre for Oral Diseases, West China Hospital of Stomatology, Sichuan University, 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:affiliation>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041</wicri:regionArea></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:affiliation>Pediatric Intensive Care Unit, Department of Critical Care Medicine, West China Hospital of Sichuan University, Chengdu, China.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Pediatric Intensive Care Unit, Department of Critical Care Medicine, West China Hospital of Sichuan University, 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:affiliation>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China. jijiyuanyuan@163.com.</nlm:affiliation><country xml:lang="fr">République populaire de Chine</country><wicri:regionArea>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041</wicri:regionArea></affiliation></author></analytic><series><title level="j">Respiratory research</title><idno type="eISSN">1465-993X</idno><imprint><date when="2020" type="published">2020</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Lymphangioleiomyomatosis (LAM) is a rare, low-grade multisystem neoplastic disease. 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.</div></front></TEI><pubmed><MedlineCitation Status="In-Process" Owner="NLM"><PMID Version="1">32059669</PMID><DateRevised><Year>2020</Year><Month>02</Month><Day>21</Day></DateRevised><Article PubModel="Electronic"><Journal><ISSN IssnType="Electronic">1465-993X</ISSN><JournalIssue CitedMedium="Internet"><Volume>21</Volume><Issue>1</Issue><PubDate><Year>2020</Year><Month>Feb</Month><Day>14</Day></PubDate></JournalIssue><Title>Respiratory research</Title><ISOAbbreviation>Respir. Res.</ISOAbbreviation></Journal><ArticleTitle>The efficacy and safety of pharmacological treatments for lymphangioleiomyomatosis.</ArticleTitle><Pagination><MedlinePgn>55</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1186/s12931-020-1316-3</ELocationID><Abstract><AbstractText Label="BACKGROUND" NlmCategory="BACKGROUND">Lymphangioleiomyomatosis (LAM) is a rare, low-grade multisystem neoplastic disease. 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.</AbstractText><AbstractText Label="METHODS" NlmCategory="METHODS">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.</AbstractText><AbstractText Label="RESULTS" NlmCategory="RESULTS">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 (P > 0.05), with the pooled response rate of AML being 0.62 (95% confidence intervals [CIs]: 0.43 to 0.82, I<sup>2</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 (P > 0.05), while the pooled response rate of AML was 0.78 (95% CI: 0.68 to 0.88, I<sup>2</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.</AbstractText><AbstractText Label="CONCLUSIONS" NlmCategory="CONCLUSIONS">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.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Wang</LastName><ForeName>Qi</ForeName><Initials>Q</Initials><AffiliationInfo><Affiliation>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Luo</LastName><ForeName>Mengqi</ForeName><Initials>M</Initials><AffiliationInfo><Affiliation>State Key Laboratory of Oral Diseases, National Clinical Research Centre for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu, China.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Xiang</LastName><ForeName>Bo</ForeName><Initials>B</Initials><AffiliationInfo><Affiliation>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Chen</LastName><ForeName>Siyuan</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>Pediatric Intensive Care Unit, Department of Critical Care Medicine, West China Hospital of Sichuan University, Chengdu, China.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Ji</LastName><ForeName>Yi</ForeName><Initials>Y</Initials><Identifier Source="ORCID">http://orcid.org/0000-0002-9289-9660</Identifier><AffiliationInfo><Affiliation>Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, #37# Guo-Xue-Xiang, Chengdu, 610041, China. jijiyuanyuan@163.com.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><GrantList CompleteYN="Y"><Grant><GrantID>81400862</GrantID><Agency>National Natural Science Foundation of China</Agency><Country></Country></Grant><Grant><GrantID>81401606</GrantID><Agency>National Natural Science Foundation of China</Agency><Country></Country></Grant><Grant><GrantID>19ZDYF1454</GrantID><Agency>the Key Project in the Science & Technology Program of Sichuan Province</Agency><Country></Country></Grant><Grant><GrantID>2015SU04A15</GrantID><Agency>Science Foundation for Excellent Youth Scholars of Sichuan University</Agency><Country></Country></Grant><Grant><GrantID>2019HXFH056</GrantID><Agency>the 1·3·5 project for disciplines of excellence-Clinical Research Incubation Project, West China Hospital, Sichuan University</Agency><Country></Country></Grant></GrantList><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2020</Year><Month>02</Month><Day>14</Day></ArticleDate></Article><MedlineJournalInfo><Country>England</Country><MedlineTA>Respir 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