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Newborn screening for Krabbe disease: the New York State model.

Authors :
Duffner PK
Caggana M
Orsini JJ
Wenger DA
Patterson MC
Crosley CJ
Kurtzberg J
Arnold GL
Escolar ML
Adams DJ
Andriola MR
Aron AM
Ciafaloni E
Djukic A
Erbe RW
Galvin-Parton P
Helton LE
Kolodny EH
Kosofsky BE
Kronn DF
Kwon JM
Levy PA
Miller-Horn J
Naidich TP
Pellegrino JE
Provenzale JM
Rothman SJ
Wasserstein MP
Source :
Pediatric neurology [Pediatr Neurol] 2009 Apr; Vol. 40 (4), pp. 245-52; discussion 253-5.
Publication Year :
2009

Abstract

Krabbe disease is a rare inherited neurologic disorder affecting the central and peripheral nervous systems. The disease has four phenotypes: early infantile, later onset, adolescent, and adult. The only known treatment is hematopoietic stem cell transplantation, which is, in the early infantile form of the disease, most beneficial if performed before onset of clinical symptoms. In August 2006, New York State began screening all newborns for Krabbe disease. A rapid and accurate technique for assessing galactocerebrosidase activity and performing DNA mutation analysis had been developed. Interpreting these results was limited, however, because neither enzyme activity nor genetic mutation reliably predicts phenotype. A series of initiatives were therefore developed by a multidisciplinary group of neurologists, geneticists, metabolic pediatricians, neurodevelopmental pediatricians, and transplant physicians (the Krabbe Consortium of New York State) to enhance the effectiveness of the newborn screening program. A standardized clinical evaluation protocol was designed based on the available literature, criteria for transplantation for the early infantile phenotype were formulated, a clinical database and registry was developed, and a study of developmental and functional outcomes was instituted. This multidisciplinary standardized approach to evaluating infants who have positive results on newborn screening may serve as a model for other states as they begin the process of screening for Krabbe disease and other lysosomal storage disorders.

Details

Language :
English
ISSN :
0887-8994
Volume :
40
Issue :
4
Database :
MEDLINE
Journal :
Pediatric neurology
Publication Type :
Academic Journal
Accession number :
19302934
Full Text :
https://doi.org/10.1016/j.pediatrneurol.2008.11.010