RESUMO
Cor triatriatum dexter is a rare congenital abnormality in which the right atrium is divided into two chambers by a membrane. A rare variant of cor triatriatum dexter where the membrane is attached to the left of the superior vena cava at one end and to the right of inferior vena cava and coronary sinus at the other end has been described only once before. We describe here a case of this very rare variant of cor triatriatum dexter that was diagnosed using contrast echocardiography.
Assuntos
Meios de Contraste , Coração Triatriado/diagnóstico , Ecocardiografia/métodos , Aumento da Imagem/métodos , Idoso , Coração Triatriado/diagnóstico por imagem , Diagnóstico Diferencial , Átrios do Coração/anormalidades , Átrios do Coração/diagnóstico por imagem , Humanos , Masculino , Doenças RarasRESUMO
PURPOSE: Mammalian target of rapamycin (mTOR) inhibitors like temsirolimus may result in undesirable AKT upregulation. Metformin inhibits mTOR through different mechanisms and may enhance temsirolimus's antitumor activity. We conducted an open-label phase I dose escalation trial of this drug combination in patients with advanced/refractory cancers. METHODS: Temsirolimus, 25 mg weekly, was combined with an escalating daily dose of metformin (level 1: 500; level 2: 1000; level 3: 1500; level 4: 2000 mg) by utilizing a standard 3 + 3 trial design. Treatment was administered in 28-day cycles following initial 2-week metformin titration during the first cycle. RESULTS: Twenty-one patients (median age, 56 years) with sarcoma (n = 8), colorectal (n = 3), endometrial (n = 4), uterine carcinosarcoma (n = 2), ovarian (n = 2), and other (n = 2) cancers were enrolled. Patients had received median of four prior systemic treatments. Two dose-limiting toxicities were observed (grade 3 mucositis, grade 3 renal failure); both patients continued treatment after dose modification. Fifty-six percent patients had stable disease as best response; clinical benefit rate was 22 %. Patients continued treatment for median of 11 weeks. CONCLUSIONS: Combination temsirolimus/metformin was well tolerated with modestly promising effectiveness among this heavily pretreated patient cohort. We recommend a dose of temsirolimus 25 mg weekly and metformin 2000 mg daily for phase II study.