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Regression of Cutaneous and Gastrointestinal Telangiectasia with Sirolimus and Aspirin in a Patient with Hereditary Hemorrhagic Telangiectasia

2006· letter· en· W1990066287 on OpenAlexaffabout
Vivian C. McAlister

Bibliographic record

VenueAnnals of Internal Medicine · 2006
Typeletter
Languageen
FieldMedicine
TopicVascular Anomalies and Treatments
Canadian institutionsUniversity Hospital
Fundersnot available
KeywordsMedicineTelangiectasiaTelangiectasesAngiodysplasiaGastroenterologyPortal hypertensionThrombotic microangiopathySirolimusLiver transplantationPathologyInternal medicineCirrhosisTransplantationDisease

Abstract

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Letters7 February 2006Regression of Cutaneous and Gastrointestinal Telangiectasia with Sirolimus and Aspirin in a Patient with Hereditary Hemorrhagic TelangiectasiaVivian C. McAlister, MDVivian C. McAlister, MDFrom University Hospital, London, Ontario N6A 5A5, Canada.Author, Article, and Disclosure Informationhttps://doi.org/10.7326/0003-4819-144-3-200602070-00030 SectionsAboutFull TextPDF ToolsAdd to favoritesDownload CitationsTrack CitationsPermissions ShareFacebookTwitterLinkedInRedditEmail TO THE EDITOR:Background: Angiodysplastic lesions, including telangiectases and arteriovenous malformations, may affect the organs of many patients with hereditary hemorrhagic telangiectasia (HHT). Abnormalities in transforming growth factor-β signaling and increased production of vascular endothelial growth factor (VEGF) are thought to be responsible for HHT (1). Mucosal hemorrhage from nasopharyngeal and gastrointestinal angiodysplasia is common and is often resistant to conventional treatment. Hepatic arteriovenous malformations may cause arteriovenous shunting within the liver, ischemic hepatobiliary damage, and high-output cardiac failure (2). Liver transplantation may correct the liver complications and heart failure and may also cause regression of cutaneous arteriovenous malformations (spider ...References1. Cirulli A, Liso A, D'Ovidio F, Mestice A, Pasculli G, Gallitelli M, et al. Vascular endothelial growth factor serum levels are elevated in patients with hereditary hemorrhagic telangiectasia. Acta Haematol. 2003;110:29-32. [PMID: 12975554] CrossrefMedlineGoogle Scholar2. Garcia-Tsao G, Korzenik JR, Young L, Henderson KJ, Jain D, Byrd B, et al. Liver disease in patients with hereditary hemorrhagic telangiectasia. N Engl J Med. 2000;343:931-6. [PMID: 11006369] CrossrefMedlineGoogle Scholar3. Stallone G, Schena A, Infante B, Di Paolo S, Loverre A, Maggio G, et al. Sirolimus for Kaposi's sarcoma in renal-transplant recipients. N Engl J Med. 2005;352:1317-23. [PMID: 15800227] CrossrefMedlineGoogle Scholar4. McAlister VC, Gao Z, Peltekian K, Domingues J, Mahalati K, MacDonald AS. Sirolimus-tacrolimus combination immunosuppression [Letter]. Lancet. 2000;355:376-7. [PMID: 10665560] CrossrefMedlineGoogle Scholar5. Rajnoch J, Lodererova A, Szabo A, Honsova E, Vannay A, Bloudickova S, et al. Regulators of angiogenesis in renal ischemia/reperfusion injury in normotensive and hypertensive rats: effect of tacrolimus. Transplant Proc. 2005;37:352-4. [PMID: 15808641] CrossrefMedlineGoogle Scholar Author, Article, and Disclosure InformationAuthors: Vivian C. McAlister, MDAffiliations: From University Hospital, London, Ontario N6A 5A5, Canada.Disclosures: None disclosed. PreviousarticleNextarticle Advertisement FiguresReferencesRelatedDetailsSee AlsoCorrection: Regression of Cutaneous and Gastrointestinal Telangiectasia with Sirolimus and Aspirin in a Patient with Hereditary Hemorrhagic Telangiectasia Metrics Cited byBrain arteriovenous malformation in hereditary hemorrhagic telangiectasia: Recent advances in cellular and molecular mechanismsPerioperative Complications and Long-Term Follow-Up of Liver Transplantation in Hemorrhagic Hereditary Telangiectasia: Report of Three Cases and Systematic ReviewEfficacy and Safety of Tacrolimus as Treatment for Bleeding Caused by Hereditary Hemorrhagic Telangiectasia: An Open-Label, Pilot StudyRethinking growth factors: the case of BMP9 during vessel maturationTranslational medicine in hereditary hemorrhagic telangiectasiaSystematic review of sirolimus in dermatological conditionsTacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic TelangiectasiaFuture treatments for hereditary hemorrhagic telangiectasiaReview of Pharmacological Strategies with Repurposed Drugs for Hereditary Hemorrhagic Telangiectasia Related BleedingImpaired SMAD1/5 Mechanotransduction and Cx37 (Connexin37) Expression Enable Pathological Vessel Enlargement and ShuntingCorrecting Smad1/5/8, mTOR, and VEGFR2 treats pathology in hereditary hemorrhagic telangiectasia modelsEfficacy and safety of sirolimus in the treatment of vascular anomalies: A systematic reviewPI3K (Phosphatidylinositol 3-Kinase) Activation and Endothelial Cell Proliferation in Patients with Hemorrhagic Hereditary Telangiectasia Type 1Vascular AnomaliesDiagnostik und Therapie des M. OslerTacrolimus rescues the signaling and gene expression signature of endothelial ALK1 loss-of-function and improves HHT vascular pathologyEndoglin and alk1 as therapeutic targets for hereditary hemorrhagic telangiectasiaCurrent and emerging pharmacotherapies for hereditary hemorrhagic telangiectasiaAnomalías vasculares superficiales: malformaciones vascularesBases moleculares y aplicaciones farmacológicas de la vía de mTOR en dermatologíaElucidation of the mTOR Pathway and Therapeutic Applications in DermatologyBMP signaling in vascular biology and dysfunctionGenetic and Molecular Basis for Hereditary Hemorrhagic TelangiectasiaAnti-angiogenic therapeutic strategies in hereditary hemorrhagic telangiectasiaLes malformations cutanées vasculaires non syndromiquesLiver Involvement in Osler-Weber-Rendu Disease (Hereditary Hemorrhagic Telangiectasia)Immunosuppressor FK506 Increases Endoglin and Activin Receptor-Like Kinase 1 Expression and Modulates Transforming Growth Factor-β1 Signaling in Endothelial CellsHereditary Hemorrhagic Telangiectasia of the Liver Complicated by Ischemic Bile Duct Necrosis and Sepsis: Case Report and Review of the LiteratureLiver involvement in hereditary hemorrhagic telangiectasia (HHT)Correction: Regression of Cutaneous and Gastrointestinal Telangiectasia with Sirolimus and Aspirin in a Patient with Hereditary Hemorrhagic Telangiectasia 7 February 2006Volume 144, Issue 3Page: 226-227KeywordsArteriovenous malformationAspirinHeart failureHemorrhageImmunosuppressivesKaposi sarcomaLiverLiver transplantationTransplantationVascular endothelial growth factor ePublished: 7 February 2006 Issue Published: 7 February 2006 Copyright & PermissionsCopyright © 2006 by American College of Physicians. 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How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.347
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.015
GPT teacher head0.261
Teacher spread0.246 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

Study designCase report
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations50
Published2006
Admission routes2
Has abstractyes

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