Visual spatial learning outcomes for clinical trials in neurofibromatosis type 1

被引：11

作者：

Ullrich, Nicole J. ^{[1
]}

Payne, Jonathan M. ^{[2
,3
]}

Walsh, Karin S. ^{[4
]}

Cutter, Gary ^{[5
]}

Packer, Roger ^{[4
]}

North, Kathryn ^{[2
,3
]}

Rey-Casserly, Celiane ^{[6
]}

机构：

[1] Boston Childrens Hosp, Dept Neurol, 300 Longwood Ave, Boston, MA 02115 USA

[2] Royal Childrens Hosp, Murdoch Childrens Res Inst, Melbourne, Vic, Australia

[3] Univ Melbourne, Dept Paediat, Melbourne, Vic, Australia

[4] Childrens Natl Hlth Syst, Ctr Neurosci & Behav Med, Washington, DC USA

[5] Univ Alabama Birmingham, Sch Publ Hlth, Birmingham, AL 35294 USA

[6] Boston Childrens Hosp, Dept Psychiat, Boston, MA USA

来源：

ANNALS OF CLINICAL AND TRANSLATIONAL NEUROLOGY | 2020年 / 7卷 / 02期

关键词：

CHILDREN; DEFICITS;

D O I：

10.1002/acn3.50976

中图分类号：

R74 [神经病学与精神病学];

学科分类号：

摘要：

Cognitive problems are common in children with neurofibromatosis type 1, representing a significant source of lifelong morbidity. Assessment of cognitive function has been challenging in the setting of clinical trials. Spatial learning deficits may be an important target for cognitive interventions. We leveraged a large, international cognitive study in affected children with NF1 treated with lovastatin to assess spatial learning using the "Arena Maze", a portable, computerized task that allows for retesting in the same environment. As with the parent study, spatial learning assessed with this task did not improve with lovastatin treatment.

引用

页码：245 / 249

页数：5

共 6 条

[1] Birth Incidence and Prevalence of Tumor-Prone Syndromes: Estimates From a UK Family Genetic Register Service [J].

Evans, D. G. ;

Howard, E. ;

Giblin, C. ;

Clancy, T. ;

Spencer, H. ;

Huson, S. M. ;

Lalloo, F. .

AMERICAN JOURNAL OF MEDICAL GENETICS PART A, 2010, 152A (02) :327-332

[2] The nature and frequency of cognitive deficits in children with neurofibromatosis type 1 [J].

Hyman, SL ;

Shores, A ;

North, KN .

NEUROLOGY, 2005, 65 (07) :1037-1044

[3] The HMG-CoA reductase inhibitor lovastatin reverses the learning and attention deficits in a mouse model of neurofibromatosis type 1 [J].

Li, WD ;

Cui, YJ ;

Kushner, SA ;

Brown, RAM ;

Jentsch, JD ;

Frankland, PW ;

Cannon, TD ;

Silva, AJ .

CURRENT BIOLOGY, 2005, 15 (21) :1961-1967

[4] Randomized placebo-controlled study of lovastatin in children with neurofibromatosis type 1 [J].

Payne, Jonathan M. ;

Barton, Belinda ;

Ullrich, Nicole J. ;

Cantor, Alan ;

Hearps, Stephen J. C. ;

Cutter, Gary ;

Rosser, Tena ;

Walsh, Karin S. ;

Gioia, Gerard A. ;

Wolters, Pamela L. ;

Tonsgard, James ;

Schorry, Elizabeth ;

Viskochil, David ;

Klesse, Laura ;

Fisher, Michael ;

Gutmann, David H. ;

Silva, Alcino J. ;

Hunter, Scott J. ;

Rey-Casserly, Celiane ;

Cantor, Nancy L. ;

Byars, Anna W. ;

Stavinoha, Peter L. ;

Ackerson, Joseph D. ;

Armstrong, Carol L. ;

Isenberg, Jill ;

O'Neil, Sharon H. ;

Packer, Roger J. ;

Korf, Bruce ;

Acosta, Maria T. ;

North, Kathryn N. .

NEUROLOGY, 2016, 87 (24) :2575-2584

[5] Paired associate learning in children with neurofibromatosis type 1: implications for clinical trials [J].

Payne, Jonathan M. ;

Barton, Belinda ;

Shores, E. Arthur ;

North, Kathryn N. .

JOURNAL OF NEUROLOGY, 2013, 260 (01) :214-220

[6] Pilot Study of a Novel Computerized Task to Assess Spatial Learning in Children and Adolescents With Neurofibromatosis Type 1 [J].

Ullrich, Nicole J. ;

Ayr, Lauren ;

Leaffer, Emily ;

Irons, Mira B. ;

Rey-Casserly, Celiane .

JOURNAL OF CHILD NEUROLOGY, 2010, 25 (10) :1195-1202

← 1 →