Literature DB >> 19302934

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

Patricia K Duffner1, Michele Caggana, Joseph J Orsini, David A Wenger, Marc C Patterson, Carl J Crosley, Joanne Kurtzberg, Georgianne L Arnold, Maria L Escolar, Darius J Adams, Mary R Andriola, Alan M Aron, Emma Ciafaloni, Alexandra Djukic, Richard W Erbe, Patricia Galvin-Parton, Laura E Helton, Edwin H Kolodny, Barry E Kosofsky, David F Kronn, Jennifer M Kwon, Paul A Levy, Jill Miller-Horn, Thomas P Naidich, Joan E Pellegrino, James M Provenzale, Stanley J Rothman, Melissa P Wasserstein.   

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.

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Year:  2009        PMID: 19302934     DOI: 10.1016/j.pediatrneurol.2008.11.010

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  57 in total

Review 1.  Newborn screening for neuropathic lysosomal storage disorders.

Authors:  Wuh-Liang Hwu; Yin-Hsiu Chien; Ni-Chung Lee
Journal:  J Inherit Metab Dis       Date:  2010-06-08       Impact factor: 4.982

2.  [Newborn screening as a predictive genetic test: principles and challenges].

Authors:  Johannes Zschocke
Journal:  Wien Med Wochenschr       Date:  2012-03-28

Review 3.  Newborn bloodspot screening policy framework for Australia.

Authors:  Peter O'Leary; Susannah Maxwell
Journal:  Australas Med J       Date:  2015-09-30

4.  Newborn Screening for Lysosomal Storage Disorders: Views of Genetic Healthcare Providers.

Authors:  Emily C Lisi; Shawn E McCandless
Journal:  J Genet Couns       Date:  2015-08-29       Impact factor: 2.537

5.  A comparative study of patients' perceptions of genetic and genomic medicine services in California and Malaysia.

Authors:  Emily Qian; Meow-Keong Thong; Pamela Flodman; Jay Gargus
Journal:  J Community Genet       Date:  2018-12-03

Review 6.  Clinical neurogenetics: neuropathic lysosomal storage disorders.

Authors:  Gregory M Pastores; Gustavo H B Maegawa
Journal:  Neurol Clin       Date:  2013-11       Impact factor: 3.806

Review 7.  Newborn screening for Krabbe's disease.

Authors:  Joseph J Orsini; Carlos A Saavedra-Matiz; Michael H Gelb; Michele Caggana
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

8.  Evidence for improved survival in postsymptomatic stem cell-transplanted patients with Krabbe's disease.

Authors:  Thomas J Langan; Amy L Barcykowski; Jonathan Dare; Erin C Pannullo; Leah Muscarella; Randy L Carter
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

9.  Measurement of psychosine in dried blood spots--a possible improvement to newborn screening programs for Krabbe disease.

Authors:  Coleman T Turgeon; Joseph J Orsini; Karen A Sanders; Mark J Magera; Thomas J Langan; Maria L Escolar; Patricia Duffner; Devin Oglesbee; Dimitar Gavrilov; Silvia Tortorelli; Piero Rinaldo; Kimiyo Raymond; Dietrich Matern
Journal:  J Inherit Metab Dis       Date:  2015-03-12       Impact factor: 4.982

10.  Characterization and application of a disease-cell model for a neurodegenerative lysosomal disease.

Authors:  Jameson J Ribbens; Ann B Moser; Walter C Hubbard; Ernesto R Bongarzone; Gustavo H B Maegawa
Journal:  Mol Genet Metab       Date:  2013-09-21       Impact factor: 4.797

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