TY - JOUR
T1 - Exploring barriers to long-term follow-up in newborn screening programs
AU - Hoff, Timothy
AU - Hoyt, Adrienne
AU - Therrell, Brad
AU - Ayoob, Maria
N1 - Copyright:
Copyright 2008 Elsevier B.V., All rights reserved.
PY - 2006/9
Y1 - 2006/9
N2 - PURPOSE: To inform current policy debate regarding the public health role in long-term follow-up for individuals with disorders identified through state newborn testing efforts, by identifying and assessing key challenges, quality assurance activities, and long-term follow-up perceptions of state newborn screening programs. METHODS: A 23-question, web-based survey sent to newborn screening programs in all 50 states and Washington, DC, during January and February, 2005. RESULTS: Many U.S. newborn screening programs do not currently engage in structured long-term follow-up for newborns with diagnosed disorders. The newborn screening programs that do long-term follow-up face various challenges that may impact their ability to perform it effectively. Barriers include a lack of comprehensive quality assurance practices, outsourcing, financial constraints, and perceived communication problems with providers who treat these patients. CONCLUSIONS: A more in-depth analysis of the desired and feasible roles that should be played by newborn screening programs in long-term follow-up is needed. The wide variability within programs regarding the structure and implementation of long-term follow-up, and the relative absence of long-term follow-up systems components in some newborn screening programs, poses an interesting question regarding responsibility for evaluation of longer-term outcomes associated with newborn testing and disorder diagnosis.
AB - PURPOSE: To inform current policy debate regarding the public health role in long-term follow-up for individuals with disorders identified through state newborn testing efforts, by identifying and assessing key challenges, quality assurance activities, and long-term follow-up perceptions of state newborn screening programs. METHODS: A 23-question, web-based survey sent to newborn screening programs in all 50 states and Washington, DC, during January and February, 2005. RESULTS: Many U.S. newborn screening programs do not currently engage in structured long-term follow-up for newborns with diagnosed disorders. The newborn screening programs that do long-term follow-up face various challenges that may impact their ability to perform it effectively. Barriers include a lack of comprehensive quality assurance practices, outsourcing, financial constraints, and perceived communication problems with providers who treat these patients. CONCLUSIONS: A more in-depth analysis of the desired and feasible roles that should be played by newborn screening programs in long-term follow-up is needed. The wide variability within programs regarding the structure and implementation of long-term follow-up, and the relative absence of long-term follow-up systems components in some newborn screening programs, poses an interesting question regarding responsibility for evaluation of longer-term outcomes associated with newborn testing and disorder diagnosis.
KW - Long-term follow-up
KW - Newborn screening
KW - Public health
KW - State policy
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U2 - 10.1097/01.gim.0000237790.54074.3d
DO - 10.1097/01.gim.0000237790.54074.3d
M3 - Article
C2 - 16980812
AN - SCOPUS:33748742728
VL - 8
SP - 563
EP - 570
JO - Genetics in Medicine
JF - Genetics in Medicine
SN - 1098-3600
IS - 9
ER -