TY - JOUR
T1 - Newborn screening programs and sickle cell disease
T2 - A public health services and systems approach
AU - Minkovitz, Cynthia S.
AU - Grason, Holly
AU - Ruderman, Marjory
AU - Casella, James F.
N1 - Funding Information:
We are most grateful to Elizabeth Harrision, who served as project coordinator and in whose memory this manuscript is dedicated. In addition, thanks are extended to the stakeholders who generously participated in surveys and interviews. We also gratefully appreciate the contributions of a national Advisory Committee to sample selection, instrument development, and interpretation of study findings. Members and their affiliations during the time of ths study included Thomas Howard, MD (Department of Pediatrics, University of Alabama at Birmingham); Christopher Kus, MD, MPH (Division of Family Health, New York State Derpartment of Health); Marie Mann, MD, MPH (Maternal and Child Health Bureau, Health Resources and Services Administration); Richard Olney, MD, MPH (National Center on Birth Defects and Developmental Disabilities, CDC); Susan Panny, MD (Office for Genetics and Children with Special Health Care Needs, Maryland Department of Health and Mental Hygiene); Lauren Ramos, MPH (Association of Maternal and Child Health Programs); Derek Robertson, MBA, JD (Consumer); and Brad Therrell, PhD (National Newborn Screening and Genetics Resource Center, University of Texas Health Science Center at San Antonio). This research was supported by grant U54HL090515 from the National Heart Lung and Blood Institute. The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIH.
Funding Information:
Publication of this article was supported by the Centers for Disease Control and Prevention.
Publisher Copyright:
© 2016 American Journal of Preventive Medicine.
PY - 2016/7/1
Y1 - 2016/7/1
N2 - Introduction Despite universal newborn screening (NBS), children in the U.S. continue to experience morbidity and mortality from sickle cell disease and related causes. Recognizing that assessments of public health services and systems can improve public health system performance and ultimately health outcomes, this study examined variations in NBS program activities for sickle cell disease. Methods A mixed methods study included (1) a 2009 survey of NBS programs based on ten essential public health services (N=39 states with ten or more sickle cell births over a 3-year period) and (2) key informant interviews in 2011 with 13 states that had sufficient Phase 1 survey scores, black births, and variability in state legislation and geography. Key informants were from 13 NBS programs, 22 sickle cell treatment centers, and ten advocacy organizations. Analyses were conducted in 2009-2014. Results Considerable variability exists across states in program activities and roles. More programs reported activities oriented to care of individuals - ensuring access to services, coordination, and provider education; fewer reported planning and analysis activities oriented to statewide policy development and system change. Numbers of activities were not related to the number of affected births. In-depth interviews identified opportunities to enhance activities that support statewide comprehensive systems of care. Conclusions NBS programs perform important public health roles that complement and enhance clinical services. Nationwide efforts are needed to enable NBS programs to strengthen population-based functions that are essential to ensuring quality of care for the entire population of children and families affected by sickle cell disease.
AB - Introduction Despite universal newborn screening (NBS), children in the U.S. continue to experience morbidity and mortality from sickle cell disease and related causes. Recognizing that assessments of public health services and systems can improve public health system performance and ultimately health outcomes, this study examined variations in NBS program activities for sickle cell disease. Methods A mixed methods study included (1) a 2009 survey of NBS programs based on ten essential public health services (N=39 states with ten or more sickle cell births over a 3-year period) and (2) key informant interviews in 2011 with 13 states that had sufficient Phase 1 survey scores, black births, and variability in state legislation and geography. Key informants were from 13 NBS programs, 22 sickle cell treatment centers, and ten advocacy organizations. Analyses were conducted in 2009-2014. Results Considerable variability exists across states in program activities and roles. More programs reported activities oriented to care of individuals - ensuring access to services, coordination, and provider education; fewer reported planning and analysis activities oriented to statewide policy development and system change. Numbers of activities were not related to the number of affected births. In-depth interviews identified opportunities to enhance activities that support statewide comprehensive systems of care. Conclusions NBS programs perform important public health roles that complement and enhance clinical services. Nationwide efforts are needed to enable NBS programs to strengthen population-based functions that are essential to ensuring quality of care for the entire population of children and families affected by sickle cell disease.
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U2 - 10.1016/j.amepre.2016.02.019
DO - 10.1016/j.amepre.2016.02.019
M3 - Article
C2 - 27320464
AN - SCOPUS:84975061685
SN - 0749-3797
VL - 51
SP - S39-S47
JO - American journal of preventive medicine
JF - American journal of preventive medicine
IS - 1
ER -