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Pediatr Blood Cancer ; 65(9): e27228, 2018 09.
Artigo em Inglês | MEDLINE | ID: mdl-29797644

RESUMO

BACKGROUND: Previous natural history studies have advanced the understanding of sickle cell disease (SCD), but generally have not included sufficient lifespan data or investigation of the role of genetics in clinical outcomes, and have often occurred before the widespread use of disease-modifying therapies, such as hydroxyurea and chronic erythrocyte transfusions. To further advance knowledge of SCD, St. Jude Children's Research Hospital established the Sickle Cell Clinical Research and Intervention Program (SCCRIP), to conduct research in a clinically evaluated cohort of individuals with SCD across their lifetime. PROCEDURES: Initiated in 2014, the SCCRIP study prospectively recruits patients diagnosed with SCD and includes retrospective and longitudinal collection of clinical, neurocognitive, geospatial, psychosocial, and health outcomes data. Biological samples are banked for future genomics and proteomics studies. The organizational structure of SCCRIP is based upon organ/system-specific working groups and is opened to the research community for partnerships. RESULTS: As of August 2017, 1,044 (92.3% of eligible) patients with SCD have enrolled in the study (860 children and 184 adults), with 11,915 person-years of observation. Population demographics included mean age at last visit of 11.3 years (range 0.7-30.1), 49.8% females, 57.7% treated with hydroxyurea, 8.5% treated with monthly transfusions, and 62.9% hemoglobin (Hb) SS or HbSB0 -thalassemia, 25.7% HbSC, 8.4% HbsB+ -Thalassemia, 1.7% HbS/HPFH, and 1.2% other. CONCLUSIONS: The SCCRIP cohort will provide a rich resource for the conduct of high impact multidisciplinary research in SCD.


Assuntos
Anemia Falciforme/mortalidade , Estudos Longitudinais , Adolescente , Adulto , Anemia Falciforme/genética , Anemia Falciforme/terapia , Bancos de Espécimes Biológicos/organização & administração , Transfusão de Sangue , Líquidos Corporais , Criança , Pré-Escolar , Progressão da Doença , Feminino , Seguimentos , Genótipo , Hemoglobinopatias/genética , Humanos , Hidroxiureia/uso terapêutico , Lactente , Consentimento Livre e Esclarecido , Longevidade , Masculino , Seleção de Pacientes , Estudos Prospectivos , Projetos de Pesquisa , Estudos de Amostragem , Estados Unidos/epidemiologia
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