Literature DB >> 27655474

Cognitive Development in Infantile-Onset Pompe Disease Under Very Early Enzyme Replacement Therapy.

Chih-Jou Lai1,2, Ting-Rong Hsu3,4, Chia-Feng Yang3, Shyi-Jou Chen5,6, Ya-Chin Chuang1,7, Dau-Ming Niu8,9.   

Abstract

Most patients with infantile-onset Pompe disease die in early infancy before beginning enzyme replacement therapy, which has made it difficult to evaluate the impact of Pompe disease on cognitive development. Patients with infantile-onset Pompe disease can survive with enzyme replacement therapy, and physicians can evaluate cognitive development in these patients. We established an effective newborn screening program with quick clinical diagnostic criteria. Cognitive and motor development were evaluated using the Bayley Scales of Infant and Toddler Development-Third Edition at 6, 12, and 24 months of age. The patients who were treated very early demonstrate normal cognitive development with no significant change in cognition during this period (P = .18 > .05). The cognitive development was positively correlated with motor development (r = 0.533, P = .011). The results indicated that very early enzyme replacement therapy could protect cognitive development in patients with infantile-onset Pompe disease up to 24 months of age.
© The Author(s) 2016.

Entities:  

Keywords:  cognitive development; enzyme replacement therapy; infantile-onset Pompe disease

Mesh:

Year:  2016        PMID: 27655474     DOI: 10.1177/0883073816665549

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  3 in total

1.  Cognitive and academic outcomes in long-term survivors of infantile-onset Pompe disease: A longitudinal follow-up.

Authors:  Gail A Spiridigliozzi; Lori A Keeling; Mihaela Stefanescu; Cindy Li; Stephanie Austin; Priya S Kishnani
Journal:  Mol Genet Metab       Date:  2017-05-01       Impact factor: 4.797

2.  Hearing characteristics of infantile-onset Pompe disease after early enzyme-replacement therapy.

Authors:  Chien-Yu Hsueh; Chii-Yuan Huang; Chia-Feng Yang; Chia-Chen Chang; Wei-Sheng Lin; Hsiu-Lien Cheng; Shang-Liang Wu; Yen-Fu Cheng; Dau-Ming Niu
Journal:  Orphanet J Rare Dis       Date:  2021-08-05       Impact factor: 4.123

3.  The First Year Experience of Newborn Screening for Pompe Disease in California.

Authors:  Hao Tang; Lisa Feuchtbaum; Stanley Sciortino; Jamie Matteson; Deepika Mathur; Tracey Bishop; Richard S Olney
Journal:  Int J Neonatal Screen       Date:  2020-02-07
  3 in total

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