TY - JOUR
T1 - The future of newborn screening for lysosomal disorders
AU - Wasserstein, Melissa P.
AU - Orsini, Joseph J.
AU - Goldenberg, Aaron
AU - Caggana, Michele
AU - Levy, Paul A.
AU - Breilyn, Margo
AU - Gelb, Michael H.
N1 - Funding Information:
ScreenPlus is supported by the Eunice Kennedy Shriver National Institute of Child Health and Human Development of the National Institutes of Health under award number 5RO1HD073292-07. It is also supported by Abeona Therapeutics, Alexion Pharmaceuticals, the Ara Parseghian Medical Research Fund, BioMarin, Cure Sanfilippo Foundation, Dana's Angels Research Trust, the Firefly Fund, Orchard Therapeutics, PassageBio, Sanofi Genzyme, Travere Therapeutics, and Ultragenyx Pharmaceutical.
Publisher Copyright:
© 2021
PY - 2021/8/24
Y1 - 2021/8/24
N2 - The goal of newborn screening is to enhance the outcome of individuals with serious, treatable disorders through early, pre-symptomatic detection. The lysosomal storage disorders (LSDs) comprise a group of more than 50 diseases with a combined frequency of approximately 1:7000. With the availability of existing and new enzyme replacement therapies, small molecule treatments and gene therapies, there is increasing interest in screening newborns for LSDs with the goal of reducing disease-related morbidity and mortality through early detection. Novel screening methods are being developed, including efforts to enhance accuracy of screening using an array of multi-tiered, genomic, statistical, and bioinformatic approaches. While NBS data for Gaucher disease, Fabry disease, Krabbe disease, MPS I, and Pompe disease has demonstrated the feasibility of widespread screening, it has also highlighted some of the complexities of screening for LSDs. These include the identification of infants with later-onset, untreatable, and uncertain phenotypes, raising interesting ethical concerns that should be addressed as part of the NBS implementation process. Taken together, these efforts will provide critical, detailed data to help guide objective, ethically sensitive decision-making about NBS for LSDs.
AB - The goal of newborn screening is to enhance the outcome of individuals with serious, treatable disorders through early, pre-symptomatic detection. The lysosomal storage disorders (LSDs) comprise a group of more than 50 diseases with a combined frequency of approximately 1:7000. With the availability of existing and new enzyme replacement therapies, small molecule treatments and gene therapies, there is increasing interest in screening newborns for LSDs with the goal of reducing disease-related morbidity and mortality through early detection. Novel screening methods are being developed, including efforts to enhance accuracy of screening using an array of multi-tiered, genomic, statistical, and bioinformatic approaches. While NBS data for Gaucher disease, Fabry disease, Krabbe disease, MPS I, and Pompe disease has demonstrated the feasibility of widespread screening, it has also highlighted some of the complexities of screening for LSDs. These include the identification of infants with later-onset, untreatable, and uncertain phenotypes, raising interesting ethical concerns that should be addressed as part of the NBS implementation process. Taken together, these efforts will provide critical, detailed data to help guide objective, ethically sensitive decision-making about NBS for LSDs.
KW - Lysosomal storage disorders
KW - Newborn screening
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U2 - 10.1016/j.neulet.2021.136080
DO - 10.1016/j.neulet.2021.136080
M3 - Article
C2 - 34166724
AN - SCOPUS:85109170671
SN - 0304-3940
VL - 760
JO - Neuroscience Letters
JF - Neuroscience Letters
M1 - 136080
ER -