R. Eldridge, Martha Bridge Denckla, Ellen Bien, Susan E. Myers, Muriel I. Kaiser-Kupfer, A. Pikus, Sandra Schlesinger, D. M. Parry, James M. Dambrosia, Michael Zasloff, John J. Mulvihill
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引用次数: 93
摘要
1型神经纤维瘤病(NF1)的神经和认知功能在一项对照试验中进行了评估,研究对象是13对6至27岁的兄弟姐妹。每对中有一名受试者受到NF1的影响,而另一名对照受试者则未受影响。排除有局灶性中枢神经系统疾病证据的受试者。13名患有NF1的受试者没有过度的智力迟钝、注意力缺陷障碍或特殊学习障碍(使用Wilcoxon's Signed Rank检验和McNemar's Test for Symmetry)。然而,与未受影响的兄弟姐妹相比,这些受试者在细微神经异常方面的得分明显较高(21比6),而全面智商得分明显较低(94比105)。受影响的受试者的智商分数并没有集中在量表的低端,但与他们的兄弟姐妹相比,在分布上有轻微的下降。此外,在接受评估的9名受影响的受试者中,有8人存在视觉空间定向缺陷。研究结果表明,患有NF1的受试者在发育过程中大脑有广泛的改变,表现为一种或多种特定类型的神经心理缺陷。
Neurofibromatosis type 1 (Recklinghausen's disease). Neurologic and cognitive assessment with sibling controls.
Neurologic and cognitive function in neurofibromatosis type 1 (NF1) were assessed in a controlled pilot study of 13 pairs of siblings aged 6 to 27 years. One subject in each pair was affected with NF1, and the other, the control subject, was unaffected. Subjects with evidence of focal central nervous system disease were excluded. The 13 subjects with NF1 had no excess of mental retardation, attention-deficit disorder, or specific learning disorders (using Wilcoxon's Signed Rank Test and McNemar's Test for Symmetry). These subjects, however, had significantly higher scores for subtle neurologic abnormalities (21 vs 6) and significantly lower full-scale IQ scores (94 vs 105) than their unaffected siblings. The IQ scores of the affected subjects were not clustered at the lower end of the scale but showed a slight downward shift in distribution compared with those of their siblings. In addition, a visual-spatial orientation deficit was present in eight of nine affected subjects so evaluated. The findings suggest that subjects with NF1 have a widespread alteration of the brain during development that manifests as one or more specific types of neuropsychologic deficits.