Use of Rivaroxaban for Prophylaxis of Superficial Venous Thrombosis in Klippel-Trenaunay- Weber Syndrome - Case Report and Systematic Review
Notice bibliographique
Résumé
Abstract INTRODUCTION: We present a case of a young man with Klippel Trenaunay Weber Syndrome (KTWS) characterized by recurrent superficial vein thrombosis (SVT) complicated by an episode of disseminated intravascular coagulation (DIC) who ultimately was maintained on prophylactic rivaroxaban to prevent recurrent thrombotic events. We performed a literature search to identify similar cases and to summarize common presenting features and attempted treatment modalities. CASE DESCRIPTION: An 18 year old man with KTWS presented with a large vascular malformation over his left chest and arm developed acute worsening SVT post sodium tetradecyl injection. Further investigation revealed a severe DIC was also present. On exam, the venous malformation and left arm were extremely tender with significant bruising. Presenting labs: Hg 24, platelet 98, bilirubin 588 (unconjugated 212), LDH 1597, PTT 32, INR 1.9, fibrinogen 1.1, Ddimer >20. He was treated with aggressive supportive care for DIC and routine deep vein thrombosis (DVT) prophylaxis with low-molecular-weight heparin (LMWH). Symptoms improved after anticoagulation was started. Ultrasound of his left arm revealed old SVT with recanulization within the venous malformation. He subsequently developed recurrent painful crises in his arm secondary to recurrent SVT. These were usually treated with short term LMWH. Imaging studies ruled out any deep vein thrombotic events. We eventually elected to commence indefinite duration anticoagulation with rivaroxaban 10mg daily with significant decrease in further acute flares. METHODS: To review how thrombotic manifestations of KTWS are managed, we performed a comprehensive literature search on PubMed using MESH terms: Klippel Trenaunay Weber Syndrome, Thrombosis, DIC and Kasabach-Merritt coagulopathy. RESULTS: We found 99 abstracts. 87 were excluded for non thrombotic complications of KTWS. 13 case reports focusing on treatment of thrombotic manifestations of KTWS were analyzed further. These included 12 case reports reviewing management of DVT in KTWS and 1 case report reviewing management of SVT in KTWS however with no DIC. 3 of these abstracts also reviewed management of coagulopathy (DIC or local intravascular coagulopathy). Of the 12 case reports, 5 described patients with deep vein thrombosis in KTWS treated with therapeutic warfarin; 4 patients improved however 1 died of cor pulmonale. An additional 5 cases of DVT in KTWS were successfully managed with therapeutic LMWH. 2 of these cases were also complicated by DIC. One case report described use of therapeutic rivaroxaban (20mg daily) to manage DVT in KTWS. This case was also complicated by DIC. There were no case reports describing the prevention of SVT in KTWS. CONCLUSION: To our knowledge this is the first case report to describe successful off-label use of prophylactic rivaroxaban to prevent further thrombotic events in a patient with KTWS. Disclosures Off Label Use: RIVAROXABAN -- PROPHYLACTIC ANTI-THROMBOSIS FOR SUPERFICIAL VEIN THROMBOSIS. Wu:Leo Pharma: Consultancy, Honoraria, Membership on an entity's Board of Directors or advisory committees; Pfizer: Consultancy, Honoraria, Membership on an entity's Board of Directors or advisory committees.
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Comment cette classification a été obtenuedéplier
Prédiction machine sur la base complète
Imitation des enseignantsNi prévalence calibrée, ni vérité terrain. Validation humaine à venir. Le volet Gemma est une étiquette directe du modèle pour chaque travail de la base, lue sur la notice réduite au titre. Le volet Codex est un classifieur appris des 10 348 étiquettes directes de Codex et calibré sur les taux pondérés de l'échantillon; les champs sans appui suffisant ne portent aucun appel Codex. Le mode candidate est l'union des deux volets; le consensus est leur intersection. Ces sorties portent le statut machine_predicted_unvalidated et ne sont pas des étiquettes humaines.
Scores du classifieur distillé par catégorie (deux têtes)
| Catégorie | Codex | Gemma |
|---|---|---|
| Métarecherche | 0,001 | 0,007 |
| Méta-épidémiologie (sens strict) | 0,001 | 0,000 |
| Méta-épidémiologie (sens large) | 0,002 | 0,002 |
| Bibliométrie | 0,007 | 0,007 |
| Études des sciences et des technologies | 0,000 | 0,001 |
| Communication savante | 0,001 | 0,001 |
| Science ouverte | 0,001 | 0,001 |
| Intégrité de la recherche | 0,001 | 0,000 |
| Charge utile insuffisante (le modèle a refusé de juger) | 0,002 | 0,000 |
Scores machine (provisoires)
Les deux têtes enseignantes du modèle étudiant, lues sur ce travail. Un score ordonne la base pour la relecture; il n'affirme jamais une catégorie, et le statut de validation accompagne chaque rangée tel quel.
Scores de référence d'un modèle non mature (critères de maturité non atteints, 7 itérations). Un score ordonne; il n'affirme jamais une catégorie.
score_only:v0-immature-baseline · tel quel depuis la passe de notation : score_only signifie que le nombre peut ordonner les travaux, et qu'aucune étiquette de catégorie n'en découleClassification
machine, non validéePrédiction automatique; un appel candidat d’une seule source (Gemma direct ou Codex distillé), pas un consensus.
Le détail, modèle par modèle et score par score, se trouve en fin de page sous « Comment cette classification a été obtenue ».