Literature DB >> 19068248

The Hunter's Hope Krabbe family database.

Patricia K Duffner1, Kabir Jalal, Randolf L Carter.   

Abstract

The objective was to identify presenting signs and symptoms, age at onset of symptoms and diagnosis, and survival in a large population of children with Krabbe disease. In 1997, Hunter's Hope Foundation began collecting clinical data on patients who had been diagnosed with Krabbe disease. As of June 2006, 334 families had returned questionnaires. Deidentified data were analyzed, including country of origin, sex, age at onset of symptoms, symptoms before diagnosis, age at diagnosis, symptoms after diagnosis, initial diagnosis, and survival. Seventy-one percent of patients developed symptoms at 0 to 6 months of age, 19% between 7 and 12 months, and 10% at 13 months + (13 months-5.5 years). The most common initial symptoms for age 0 to 12 months were crying and irritability, stiffness, and seizures. Older children were more likely to present with gait disturbances or loss of milestones. Survival differed according to age at onset of symptoms. Children with the early infantile phenotype (onset 0 to 6 months) had significantly worse survivals than either those with onset at 7 to 12 months or at 13 months to 5.5 years. Given that neither galactocerebrosidase activity nor mutation analysis reliably predict disease severity, the data from this study should help investigators recognize the earliest symptoms of the disease, as well as increase awareness of age of onset and natural history of the various phenotypes.

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Year:  2009        PMID: 19068248     DOI: 10.1016/j.pediatrneurol.2008.08.011

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


  20 in total

Review 1.  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

2.  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

3.  Clinical, electrophysiological, and biochemical markers of peripheral and central nervous system disease in canine globoid cell leukodystrophy (Krabbe's disease).

Authors:  Allison M Bradbury; Jessica H Bagel; Xuntian Jiang; Gary P Swain; Maria L Prociuk; Caitlin A Fitzgerald; Patricia A O'Donnell; Kyle G Braund; Daniel S Ory; Charles H Vite
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

Review 4.  A microglial hypothesis of globoid cell leukodystrophy pathology.

Authors:  Alexandra M Nicaise; Ernesto R Bongarzone; Stephen J Crocker
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

5.  Quantitative Microproteomics Based Characterization of the Central and Peripheral Nervous System of a Mouse Model of Krabbe Disease.

Authors:  Davide Pellegrini; Ambra Del Grosso; Lucia Angella; Nadia Giordano; Marialaura Dilillo; Ilaria Tonazzini; Matteo Caleo; Marco Cecchini; Liam A McDonnell
Journal:  Mol Cell Proteomics       Date:  2019-03-29       Impact factor: 5.911

6.  Newborn screening for lysosomal storage diseases: an ethical and policy analysis.

Authors:  Lainie Friedman Ross
Journal:  J Inherit Metab Dis       Date:  2011-12-22       Impact factor: 4.982

7.  An in vitro model for the study of cellular pathophysiology in globoid cell leukodystrophy.

Authors:  Kumiko I Claycomb; Kasey M Johnson; Ernesto R Bongarzone; Stephen J Crocker
Journal:  J Vis Exp       Date:  2014-10-21       Impact factor: 1.355

8.  The burden of inherited leukodystrophies in children.

Authors:  J L Bonkowsky; C Nelson; J L Kingston; F M Filloux; M B Mundorff; R Srivastava
Journal:  Neurology       Date:  2010-07-21       Impact factor: 9.910

9.  Relative incidence of inherited white matter disorders in childhood to acquired pediatric demyelinating disorders.

Authors:  Adeline Vanderver; Heather Hussey; Johanna L Schmidt; William Pastor; Heather J Hoffman
Journal:  Semin Pediatr Neurol       Date:  2012-12       Impact factor: 1.636

10.  Aberrant production of tenascin-C in globoid cell leukodystrophy alters psychosine-induced microglial functions.

Authors:  Kumiko I Claycomb; Paige N Winokur; Kasey M Johnson; Alexandra M Nicaise; Anthony W Giampetruzzi; Anthony V Sacino; Evan Y Snyder; Elisa Barbarese; Ernesto R Bongarzone; Stephen J Crocker
Journal:  J Neuropathol Exp Neurol       Date:  2014-10       Impact factor: 3.685

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