Saccadic eye movement characteristics in adult Niemann-Pick Type C disease: relationships with disease severity and brain structural measures.
Saccadic eye movement characteristics in adult Niemann-Pick Type C disease: relationships with disease severity and brain structural measures.
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DOI:
10.1371/journal.pone.0050947
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发表时间:
2012
期刊:
影响因子:
3.7
通讯作者:
Walterfang M
中科院分区:
文献类型:
--
作者:
Abel LA;Bowman EA;Velakoulis D;Fahey MC;Desmond P;Macfarlane MD;Looi JC;Adamson CL;Walterfang M
Niemann-Pick Type C disease (NPC) is a rare genetic disorder of lipid metabolism. A parameter related to horizontal saccadic peak velocity was one of the primary outcome measures in the clinical trial assessing miglustat as a treatment for NPC. Neuropathology is widespread in NPC, however, and could be expected to affect other saccadic parameters. We compared horizontal saccadic velocity, latency, gain, antisaccade error percentage and self-paced saccade generation in 9 adult NPC patients to data from 10 age-matched controls. These saccadic measures were correlated with appropriate MRI-derived brain structural measures (e.g., dorsolateral prefrontal cortex, frontal eye fields, supplemental eye fields, parietal eye fields, pons, midbrain and cerebellar vermis) and with measures of disease severity and duration. The best discriminators between groups were reflexive saccade gain and the two volitional saccade measures. Gain was also the strongest correlate with disease severity and duration. Most of the saccadic measures showed strongly significant correlations with neurophysiologically appropriate brain regions. While our patient sample is small, the apparent specificity of these relationships suggests that as new diagnostic methods and treatments become available for NPC, a broader range of saccadic measures may be useful tools for the assessment of disease progression and treatment efficacy.
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影响因子:
--
作者:
CURRIE, J;RAMSDEN, B;MARUFF, P
通讯作者:
MARUFF, P
影响因子:
4.4
作者:
Iturriaga, C.;Pineda, M.;Coll, M. J.
通讯作者:
Coll, M. J.
影响因子:
14.5
作者:
Pierrot-Deseilligny, C;Müri, RM;Rivaud-Pechoux, S
通讯作者:
Rivaud-Pechoux, S
影响因子:
5.7
作者:
Diedrichsen, Joern;Balsters, Joshua H.;Ramnani, Narender
通讯作者:
Ramnani, Narender
DOI:
10.1196/annals.1325.023
发表时间:
2005-01-01
期刊:
CLINICAL AND BASIC OCULOMOTOR RESEARCH: IN HONOR OF DAVID S. ZEE
影响因子:
--
作者:
Pierrot-Deseilligny, C;Müri, RM;Milea, D
通讯作者:
Milea, D