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W2087615298 abstract "Letters1 May 2007Sirolimus Treatment for Pulmonary LymphangioleiomyomatosisCamille Taillé, MD, PhD, Marie-Pierre Debray, MD, and Bruno Crestani, PhDCamille Taillé, MD, PhDFrom Hôpital Bichat–Claude Bernard and Hôpitaux de Paris, Paris 75018, France.Search for more papers by this author, Marie-Pierre Debray, MDFrom Hôpital Bichat–Claude Bernard and Hôpitaux de Paris, Paris 75018, France.Search for more papers by this author, and Bruno Crestani, PhDFrom Hôpital Bichat–Claude Bernard and Hôpitaux de Paris, Paris 75018, France.Search for more papers by this authorAuthor, Article, and Disclosure Informationhttps://doi.org/10.7326/0003-4819-146-9-200705010-00022 SectionsAboutFull TextPDF ToolsAdd to favoritesDownload CitationsTrack CitationsPermissions ShareFacebookTwitterLinkedInRedditEmail Background: Lymphangioleiomyomatosis (LAM) is a rare disease characterized by abnormal proliferation of smooth-muscle cells within the lung (1) that are responsible for the cystic destruction of lung parenchyma, leading to chronic respiratory failure (2). No effective treatment, except for lung transplantation, is available. Abdominal tumors (renal angiomyolipomas, lymphangioleiomyomas, and enlarged lymph nodes) are frequently associated with lung involvement. Recently, loss-of-function mutations in the tumor suppressor genes tuberous sclerosis 1 and 2 (TSC1 and TSC2, respectively) have been described in LAM (3). In vitro studies on LAM smooth-muscle cells have shown that mutation on the TSC1–TSC2 complex dysregulates the activation of ...References1. Finlay G. The LAM cell: what is it, where does it come from, and why does it grow? [Editorial]. Am J Physiol Lung Cell Mol Physiol. 2004;286:L690-3. [PMID: 15003933] CrossrefMedlineGoogle Scholar2. Sullivan EJ. Lymphangioleiomyomatosis: a review. Chest. 1998;114:1689-703. [PMID: 9872207] CrossrefMedlineGoogle Scholar3. Strizheva GD, Carsillo T, Kruger WD, Sullivan EJ, Ryu JH, Henske EP. The spectrum of mutations in TSC1 and TSC2 in women with tuberous sclerosis and lymphangiomyomatosis. Am J Respir Crit Care Med. 2001;163:253-8. [PMID: 11208653] CrossrefMedlineGoogle Scholar4. Goncharova EA, Goncharov DA, Eszterhas A, Hunter DS, Glassberg MK, Yeung RS, et al. Tuberin regulates p70 S6 kinase activation and ribosomal protein S6 phosphorylation. A role for the TSC2 tumor suppressor gene in pulmonary lymphangioleiomyomatosis (LAM). J Biol Chem. 2002;277:30958-67. [PMID: 12045200] CrossrefMedlineGoogle Scholar5. Goncharova EA, Goncharov DA, Spaits M, Noonan DJ, Talovskaya E, Eszterhas A, et al. Abnormal growth of smooth muscle-like cells in lymphangioleiomyomatosis: role for tumor suppressor TSC2. Am J Respir Cell Mol Biol. 2006;34:561-72. [PMID: 16424383] CrossrefMedlineGoogle Scholar Author, Article, and Disclosure InformationAffiliations: From Hôpital Bichat–Claude Bernard and Hôpitaux de Paris, Paris 75018, France.Disclosures: None disclosed. PreviousarticleNextarticle Advertisement FiguresReferencesRelatedDetails Metrics Cited ByEfficacy of Sirolimus Treatment in PEComa–10 Years of Practice PerspectivePEComas: An Uncommon Family of Sarcomas Sensitive to Targeted TherapyThe UK guidelines for management and surveillance of Tuberous Sclerosis ComplexLoss of Leucyl-tRNA synthetase b leads to ILFS1-like symptoms in zebrafishThe potential of emerging new therapeutics for the treatment of perivascular epithelioid cell tumors (PEComa)PEComa: morphology and genetics of a complex tumor familyRecurrent Disease after Lung TransplantationEverolimus treatment of abdominal lymphangioleiomyoma in five women with sporadic lymphangioleiomyomatosisEfficacy and safety of everolimus for subependymal giant cell astrocytomas associated with tuberous sclerosis complex (EXIST-1): a multicentre, randomised, placebo-controlled phase 3 trialMultimodality therapy for metastatic sarcomas confined to the lungOptimizing treatments for lymphangioleiomyomatosisIs sirolimus a therapeutic option for patients with progressive pulmonary lymphangioleiomyomatosis?A topical combination of rapamycin and tacrolimus for the treatment of angiofibroma due to tuberous sclerosis complex (TSC): a pilot study of nine Japanese patients with TSC of different disease severityCurrent management of lymphangioleiomyomatosisAdvances in sarcoma genomics and new therapeutic targetsChanges in Lung Function and Chylous Effusions in Patients With Lymphangioleiomyomatosis Treated With SirolimusAngelo M. Taveira-DaSilva, MD, PhD, Olanda Hathaway, CRNP, Mario Stylianou, PhD, and Joel Moss, MD, PhDResponse of Lymphangioleiomyomatosis to a Mammalian Target of Rapamycin Inhibitor (Temsirolimus) –Based TreatmentEverolimus for Subependymal Giant-Cell Astrocytomas in Tuberous SclerosisLymphangioléiomyomatoseLymphangioleiomyomatosis: A Disease Involving the Lymphatic SystemClinical Activity of mTOR Inhibition With Sirolimus in Malignant Perivascular Epithelioid Cell Tumors: Targeting the Pathogenic Activation of mTORC1 in TumorsLymphangioleiomyomatosisSirolimus (Rapamune®), a Prototype Drug of Rapamycin.Molecular Pathology of Lymphangioleiomyomatosis and Other Perivascular Epithelioid Cell TumorsRapamycin as a Therapy of Choice After Renal Transplantation in a Patient With Tuberous Sclerosis ComplexPneumopathies interstitielles diffusesCathepsin-k expression in pulmonary lymphangioleiomyomatosisSirolimus Treatment for Recurrent Lymphangioleiomyomatosis After Lung TransplantationReview: Therapies for interstitial lung disease: past, present and futureLymphangioléiomyomatoseLymphangioleiomyomatosis and Tuberous Sclerosis ComplexThe mTOR inhibitor rapamycin significantly improves facial angiofibroma lesions in a patient with tuberous sclerosisCurrent Management of Tuberous Sclerosis Complex 1 May 2007Volume 146, Issue 9Page: 687-688KeywordsCell proliferationCellsComputed axial tomographyLungsMagnetic resonance imagingMutationRespiratory failureWeaningWeight gain ePublished: 1 May 2007 Issue Published: 1 May 2007 CopyrightCopyright © 2007 by American College of Physicians. 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