Elucidating the nature and mechanism of tic improvement in tourette syndrome: a pilot study.
Elucidating the nature and mechanism of tic improvement in tourette syndrome: a pilot study.
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DOI：
10.7916/d8th8jqq
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发表时间：
2014
期刊：
影响因子：
--
通讯作者：
Anderson JS
中科院分区：
文献类型：
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作者：
Shprecher DR;Gannon K;Agarwal N;Shi X;Anderson JS
由于不明原因，许多抽动秽语综合症 (TS) 儿童报告说，在成年早期抽动几乎完全缓解。在患有 TS 的青少年和成人中，通过静息态功能 MRI (rs-fc-MRI) 观察到，大脑网络的不成熟可能会在抽动改善或缓解的成年人中演变成成熟的模式。我们探索了在我们的年轻成年 TS 男性群体中测试这一假设的可行性，每个人都在儿童时期完成了先前的临床评估，作为单独的 TS 协会遗传学联盟研究的一部分。共有 10 名 TS 男性（停用抽动抑制药物至少 6 个月），年龄 19-32 岁，平均随访间隔 7.5（2 至 13）年，以及 11 名神经系统正常对照者入组，并接受了 3-Tesla 结构和 rs-fc-MRI 序列。耶鲁全球抽动严重程度量表 (YGTSS) 的平均变化为 -31.5%（总计）和 -26.6%（YGTSS 运动+声音）。两名受试者报告抽动相关残疾得到缓解，平均分从 45 下降至 16.5（YGTSS 总分），从 25 下降至 11.5（YGTSS 运动+声音）。 Rs-fc-MRI显示同侧前扣带皮层和中扣带皮层与纹状体之间的连接性显着增加，局部连接之间的连接性增加，而更远的连接之间的连接性减少；代表不成熟的连接模式。与之前的报告类似，我们发现成人 TS 受试者的功能连接模式不成熟。尽管抽动未完全缓解，两名受试者的抽动严重程度却急剧下降，这与抽动相关的残疾改善相关。需要做更多的工作来阐明 TS 显着改善的机制。
For unclear reasons, many Tourette syndrome (TS) children report near-complete tic remission by young adulthood. Immature maturation of brain networks, observed with resting-state functional MRI (rs-fc-MRI) in adolescents and adults with TS, might evolve to a mature pattern in adults who experience tic improvement or remission. We explored the feasibility of testing this hypothesis in our population of young adult TS males, each with prior clinical assessments completed during childhood as part of a separate TS Association Genetics Consortium study. A total of 10 TS males (off tic suppressing drugs for at least 6 months) aged 19–32 years, mean follow-up interval 7.5 (2 to 13) years, and 11 neurologically normal controls were enrolled and underwent 3-Tesla structural and rs-fc-MRI sequences. The mean change in Yale Global Tic Severity Scale (YGTSS) was −31.5% (total) and −26.6% (YGTSS motor+vocal). Two subjects reported resolution of tic-related disability, with drops from mean 45 to 16.5 (YGTSS-total) and 25 to 11.5 (YGTSS motor+vocal.). Rs-fc-MRI revealed significantly increased connectivity between the ipsilateral anterior and mid cingulate cortex and striatum, increased connectivity between local connections, and decreased connectivity between more distant connections; representing an immature connectivity pattern. Similar to previous reports, we found immature patterns of functional connectivity in adult TS subjects. Despite a lack of complete tic remission, two subjects exhibited dramatic drops in tic severity that correlated with tic-related disability improvement. More work is needed to elucidate the mechanism of such dramatic improvement in TS.