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Therapeutic Effect of Sirolimus for Lymphangioleiomyomatosis Remaining in the Abdominopelvic Region After Lung Transplantation: A Case Report.

Identifieur interne : 007915 ( PubMed/Checkpoint ); précédent : 007914; suivant : 007916

Therapeutic Effect of Sirolimus for Lymphangioleiomyomatosis Remaining in the Abdominopelvic Region After Lung Transplantation: A Case Report.

Auteurs : T. Ito [Japon] ; M. Suno [Japon] ; K. Sakamoto [Japon] ; Y. Yoshizaki [Japon] ; K. Yamamoto [Japon] ; R. Nakanishi [Japon] ; Y. Hirano [Japon] ; M. Irie [Japon] ; T. Kurosaki [Japon] ; S. Otani [Japon] ; M. Yamane [Japon] ; S. Sugimoto [Japon] ; K. Miyoshi [Japon] ; T. Oto [Japon]

Source :

RBID : pubmed:26915883

Descripteurs français

English descriptors

Abstract

Sirolimus (SRL) is used to treat pulmonary lymphangioleiomyomatosis (P-LAM). There is limited evidence that SRL has systemic efficacy for the patients with extrapulmonary lymphangioleiomyomatosis (E-LAM) remaining after lung transplantation (LT) for P-LAM. This report examines the efficacy of SRL treatment for the patient with E-LAM remaining after an LT for P-LAM.

DOI: 10.1016/j.transproceed.2015.12.021
PubMed: 26915883


Affiliations:


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pubmed:26915883

Le document en format XML

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<div type="abstract" xml:lang="en">Sirolimus (SRL) is used to treat pulmonary lymphangioleiomyomatosis (P-LAM). There is limited evidence that SRL has systemic efficacy for the patients with extrapulmonary lymphangioleiomyomatosis (E-LAM) remaining after lung transplantation (LT) for P-LAM. This report examines the efficacy of SRL treatment for the patient with E-LAM remaining after an LT for P-LAM.</div>
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</JournalIssue>
<Title>Transplantation proceedings</Title>
<ISOAbbreviation>Transplant. Proc.</ISOAbbreviation>
</Journal>
<ArticleTitle>Therapeutic Effect of Sirolimus for Lymphangioleiomyomatosis Remaining in the Abdominopelvic Region After Lung Transplantation: A Case Report.</ArticleTitle>
<Pagination>
<MedlinePgn>271-4</MedlinePgn>
</Pagination>
<ELocationID EIdType="doi" ValidYN="Y">10.1016/j.transproceed.2015.12.021</ELocationID>
<ELocationID EIdType="pii" ValidYN="Y">S0041-1345(15)01208-7</ELocationID>
<Abstract>
<AbstractText Label="PURPOSE" NlmCategory="OBJECTIVE">Sirolimus (SRL) is used to treat pulmonary lymphangioleiomyomatosis (P-LAM). There is limited evidence that SRL has systemic efficacy for the patients with extrapulmonary lymphangioleiomyomatosis (E-LAM) remaining after lung transplantation (LT) for P-LAM. This report examines the efficacy of SRL treatment for the patient with E-LAM remaining after an LT for P-LAM.</AbstractText>
<AbstractText Label="CASE SUMMARY" NlmCategory="METHODS">The course of the patient's recovery from an LT for P-LAM was complicated by lymphedema in the left femoral region that was caused by two E-LAM lesions remaining in the left pelvic cavity and in the retroperitoneal area. After the LT was performed, the patient started SRL treatment to reduce the E-LAM lesions. The daily SRL dose, selected based on the standard SRL dose for P-LAM, was initiated at 1 mg/d and was maintained at 2 mg/d. The remaining E-LAM lesions and lymphedema in the left femoral region improved in approximately 9 months after the LT with the administration of both SRL and the standard immunosuppressive therapy used by Okayama University Hospital, including tacrolimus, mycophenolate mofetil, and prednisolone. The SRL and tacrolimus trough concentrations in whole blood were maintained within the therapeutic window for the next 1.5 years after initiation of SRL treatment. The patient experienced no severe adverse events that required discontinuation of the SRL treatment during this time.</AbstractText>
<AbstractText Label="CONCLUSION" NlmCategory="CONCLUSIONS">The patients with remaining E-LAM lesions may receive SRL treatment to improve the quality of life after LT for P-LAM as effective therapy in cases where the patient's recovery is complicated by E-LAM lesions.</AbstractText>
<CopyrightInformation>Copyright © 2016 Elsevier Inc. All rights reserved.</CopyrightInformation>
</Abstract>
<AuthorList CompleteYN="Y">
<Author ValidYN="Y">
<LastName>Ito</LastName>
<ForeName>T</ForeName>
<Initials>T</Initials>
<AffiliationInfo>
<Affiliation>Division of Oncology Pharmaceutical Care & Science, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Suno</LastName>
<ForeName>M</ForeName>
<Initials>M</Initials>
<AffiliationInfo>
<Affiliation>Division of Oncology Pharmaceutical Care & Science, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Japan. Electronic address: sunoma@pharm.okayama-u.ac.jp.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Sakamoto</LastName>
<ForeName>K</ForeName>
<Initials>K</Initials>
<AffiliationInfo>
<Affiliation>Division of Oncology Pharmaceutical Care & Science, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Yoshizaki</LastName>
<ForeName>Y</ForeName>
<Initials>Y</Initials>
<AffiliationInfo>
<Affiliation>Division of Oncology Pharmaceutical Care & Science, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Yamamoto</LastName>
<ForeName>K</ForeName>
<Initials>K</Initials>
<AffiliationInfo>
<Affiliation>Division of Oncology Pharmaceutical Care & Science, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Nakanishi</LastName>
<ForeName>R</ForeName>
<Initials>R</Initials>
<AffiliationInfo>
<Affiliation>Division of Oncology Pharmaceutical Care & Science, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Hirano</LastName>
<ForeName>Y</ForeName>
<Initials>Y</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Irie</LastName>
<ForeName>M</ForeName>
<Initials>M</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Kurosaki</LastName>
<ForeName>T</ForeName>
<Initials>T</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Otani</LastName>
<ForeName>S</ForeName>
<Initials>S</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Yamane</LastName>
<ForeName>M</ForeName>
<Initials>M</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Sugimoto</LastName>
<ForeName>S</ForeName>
<Initials>S</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Miyoshi</LastName>
<ForeName>K</ForeName>
<Initials>K</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y">
<LastName>Oto</LastName>
<ForeName>T</ForeName>
<Initials>T</Initials>
<AffiliationInfo>
<Affiliation>Department of Thoracic Surgery, Okayama University Hospital, Okayama, Japan.</Affiliation>
</AffiliationInfo>
</Author>
</AuthorList>
<Language>eng</Language>
<PublicationTypeList>
<PublicationType UI="D002363">Case Reports</PublicationType>
<PublicationType UI="D016428">Journal Article</PublicationType>
</PublicationTypeList>
</Article>
<MedlineJournalInfo>
<Country>United States</Country>
<MedlineTA>Transplant Proc</MedlineTA>
<NlmUniqueID>0243532</NlmUniqueID>
<ISSNLinking>0041-1345</ISSNLinking>
</MedlineJournalInfo>
<ChemicalList>
<Chemical>
<RegistryNumber>0</RegistryNumber>
<NameOfSubstance UI="D007166">Immunosuppressive Agents</NameOfSubstance>
</Chemical>
<Chemical>
<RegistryNumber>9PHQ9Y1OLM</RegistryNumber>
<NameOfSubstance UI="D011239">Prednisolone</NameOfSubstance>
</Chemical>
<Chemical>
<RegistryNumber>HU9DX48N0T</RegistryNumber>
<NameOfSubstance UI="D009173">Mycophenolic Acid</NameOfSubstance>
</Chemical>
<Chemical>
<RegistryNumber>W36ZG6FT64</RegistryNumber>
<NameOfSubstance UI="D020123">Sirolimus</NameOfSubstance>
</Chemical>
<Chemical>
<RegistryNumber>WM0HAQ4WNM</RegistryNumber>
<NameOfSubstance UI="D016559">Tacrolimus</NameOfSubstance>
</Chemical>
</ChemicalList>
<CitationSubset>IM</CitationSubset>
<MeshHeadingList>
<MeshHeading>
<DescriptorName UI="D000005" MajorTopicYN="N">Abdomen</DescriptorName>
<QualifierName UI="Q000473" MajorTopicYN="N">pathology</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D000328" MajorTopicYN="N">Adult</DescriptorName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D007165" MajorTopicYN="N">Immunosuppression</DescriptorName>
<QualifierName UI="Q000379" MajorTopicYN="N">methods</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D007166" MajorTopicYN="N">Immunosuppressive Agents</DescriptorName>
<QualifierName UI="Q000627" MajorTopicYN="Y">therapeutic use</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D008175" MajorTopicYN="N">Lung Neoplasms</DescriptorName>
<QualifierName UI="Q000188" MajorTopicYN="Y">drug therapy</QualifierName>
<QualifierName UI="Q000473" MajorTopicYN="N">pathology</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D016040" MajorTopicYN="Y">Lung Transplantation</DescriptorName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D018192" MajorTopicYN="N">Lymphangioleiomyomatosis</DescriptorName>
<QualifierName UI="Q000188" MajorTopicYN="Y">drug therapy</QualifierName>
<QualifierName UI="Q000473" MajorTopicYN="N">pathology</QualifierName>
<QualifierName UI="Q000601" MajorTopicYN="N">surgery</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D008209" MajorTopicYN="N">Lymphedema</DescriptorName>
<QualifierName UI="Q000188" MajorTopicYN="N">drug therapy</QualifierName>
<QualifierName UI="Q000209" MajorTopicYN="N">etiology</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D009173" MajorTopicYN="N">Mycophenolic Acid</DescriptorName>
<QualifierName UI="Q000031" MajorTopicYN="N">analogs & derivatives</QualifierName>
<QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D018365" MajorTopicYN="N">Neoplasm, Residual</DescriptorName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D010388" MajorTopicYN="N">Pelvis</DescriptorName>
<QualifierName UI="Q000473" MajorTopicYN="N">pathology</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D011239" MajorTopicYN="N">Prednisolone</DescriptorName>
<QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D011788" MajorTopicYN="N">Quality of Life</DescriptorName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D020123" MajorTopicYN="N">Sirolimus</DescriptorName>
<QualifierName UI="Q000627" MajorTopicYN="Y">therapeutic use</QualifierName>
</MeshHeading>
<MeshHeading>
<DescriptorName UI="D016559" MajorTopicYN="N">Tacrolimus</DescriptorName>
<QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName>
</MeshHeading>
</MeshHeadingList>
</MedlineCitation>
<PubmedData>
<History>
<PubMedPubDate PubStatus="received">
<Year>2015</Year>
<Month>10</Month>
<Day>29</Day>
</PubMedPubDate>
<PubMedPubDate PubStatus="accepted">
<Year>2015</Year>
<Month>12</Month>
<Day>22</Day>
</PubMedPubDate>
<PubMedPubDate PubStatus="entrez">
<Year>2016</Year>
<Month>2</Month>
<Day>27</Day>
<Hour>6</Hour>
<Minute>0</Minute>
</PubMedPubDate>
<PubMedPubDate PubStatus="pubmed">
<Year>2016</Year>
<Month>2</Month>
<Day>27</Day>
<Hour>6</Hour>
<Minute>0</Minute>
</PubMedPubDate>
<PubMedPubDate PubStatus="medline">
<Year>2016</Year>
<Month>11</Month>
<Day>5</Day>
<Hour>6</Hour>
<Minute>0</Minute>
</PubMedPubDate>
</History>
<PublicationStatus>ppublish</PublicationStatus>
<ArticleIdList>
<ArticleId IdType="pubmed">26915883</ArticleId>
<ArticleId IdType="pii">S0041-1345(15)01208-7</ArticleId>
<ArticleId IdType="doi">10.1016/j.transproceed.2015.12.021</ArticleId>
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</pubmed>
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<list>
<country>
<li>Japon</li>
</country>
</list>
<tree>
<country name="Japon">
<noRegion>
<name sortKey="Ito, T" sort="Ito, T" uniqKey="Ito T" first="T" last="Ito">T. Ito</name>
</noRegion>
<name sortKey="Hirano, Y" sort="Hirano, Y" uniqKey="Hirano Y" first="Y" last="Hirano">Y. Hirano</name>
<name sortKey="Irie, M" sort="Irie, M" uniqKey="Irie M" first="M" last="Irie">M. Irie</name>
<name sortKey="Kurosaki, T" sort="Kurosaki, T" uniqKey="Kurosaki T" first="T" last="Kurosaki">T. Kurosaki</name>
<name sortKey="Miyoshi, K" sort="Miyoshi, K" uniqKey="Miyoshi K" first="K" last="Miyoshi">K. Miyoshi</name>
<name sortKey="Nakanishi, R" sort="Nakanishi, R" uniqKey="Nakanishi R" first="R" last="Nakanishi">R. Nakanishi</name>
<name sortKey="Otani, S" sort="Otani, S" uniqKey="Otani S" first="S" last="Otani">S. Otani</name>
<name sortKey="Oto, T" sort="Oto, T" uniqKey="Oto T" first="T" last="Oto">T. Oto</name>
<name sortKey="Sakamoto, K" sort="Sakamoto, K" uniqKey="Sakamoto K" first="K" last="Sakamoto">K. Sakamoto</name>
<name sortKey="Sugimoto, S" sort="Sugimoto, S" uniqKey="Sugimoto S" first="S" last="Sugimoto">S. Sugimoto</name>
<name sortKey="Suno, M" sort="Suno, M" uniqKey="Suno M" first="M" last="Suno">M. Suno</name>
<name sortKey="Yamamoto, K" sort="Yamamoto, K" uniqKey="Yamamoto K" first="K" last="Yamamoto">K. Yamamoto</name>
<name sortKey="Yamane, M" sort="Yamane, M" uniqKey="Yamane M" first="M" last="Yamane">M. Yamane</name>
<name sortKey="Yoshizaki, Y" sort="Yoshizaki, Y" uniqKey="Yoshizaki Y" first="Y" last="Yoshizaki">Y. Yoshizaki</name>
</country>
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