發展性忽略於單側偏癱腦性麻痺孩童：工具發展、機制與療效驗證

Abstract

背景與目的 單側偏癱的腦性麻痺孩童在日常生活中時常會忽略其患側手，即使患側手有能力能夠協助任務，這個現象被稱為發展性忽略。減少發展性忽略是復健治療的一大目標，藉由瞭解這個現象背後可能的機制可以幫助我們選擇或設計精準的治療方案。本論文分別由三個面向來深入探討發展性忽略，包含評估工具、機制與介入方案。研究目的分別為：（一）驗證改良版 Observation Test of Capacity, Performance, and Developmental Disregard (OTCPDD) 之效度與信度；（二）根據兩個觀點來尋找發展性忽略的相關因子，分別是患側手不好使用與不知道如何使用患側手，相關因子包含感覺動作、視覺空間注意力與對於雙手活動之熟悉程度；（三）探討現存之神經復健方案，侷限誘發療法與雙側密集訓練，是否有助於減少發展性忽略的現象。 研究方法 研究一共招募26位單側偏癱的腦性麻痺孩童來驗證 Observation Test of Capacity, Performance, and Developmental Disregard-Revised (OTCPDD-R) 的效度與信度，包含建構效度、施測者間與施測者內信度。研究二共包含三個部分來探討發展性忽略的相關因子，第一個部分共招募33位單側偏癱的腦性麻痺孩童，並接受Melbourne Assessment 2 (MA2)、Semmes–Weinstein monofilaments (SWM)、Two-point discrimination (TPD) 、Stereognosis 與 OTCPDD-R 測驗。第二部分共招募16位單側偏癱的腦性麻痺孩童與17位正常發展孩童，兩組孩童皆接受兩個視覺搜尋測驗與 OTCPDD-R 測驗。第三部分共招募10位單側偏癱的腦性麻痺孩童與12位正常發展孩童，兩組孩童皆接受 OTCPDD-R 測驗，接著在孩童進行動作想像時測量腦波。研究三共招募32位單側偏癱的腦性麻痺孩童參與夏令營形式的侷限誘發療法與雙側密集訓練，兩組治療總時數皆為36小時，並於治療前與後進行 OTCPDD-R 測驗。 結果 OTCPDD-R 具有良好的建構效度、施測者內與施測者間信度。在探討發展性忽略的相關因子系列研究裡，我們發現發展性忽略與基本的動作能力、觸覺註冊與空間觸覺知覺有相關，當這些能力越低，則發展性忽略的嚴重度則越高。在空間注意力的部分，在校正掉動作損傷後，單側偏癱的腦性麻痺孩童在兩個視覺搜尋測驗中的表現與正常發展孩童無顯著差異，而發展性忽略分數與患側邊的敏感度（區辨刺激物與非刺激物的能力，d-prime）有顯著相關，尤其是在分心物較多情境下，當分心物越多的時候，孩童區辨刺激物與非刺激物的能力則越低。在腦波實驗中，只使用患側手來從事雙手任務對於單側偏癱的腦性麻痺孩童來說較為熟悉，且當發展性忽略的嚴重度越高時，在想像雙手從事任務時，患側腦的事件相關去同步化 (event-related desynchronization) 相對健側腦來的低。在介入研究中，發展性忽略只有在雙側密集訓練後有顯著的進步。 結論 OTCPDD-R 能夠用來評量單側偏癱腦性麻痺孩童的發展性忽略，且具有良好的效度與信度。本論文透過系列研究試圖去解釋發展性忽略背後的機制，根據兩個觀點（患側手不好用與不知道如何使用患側手）所提出的相關因子皆與發展性忽略有顯著相關，此結果初步提供造成發展性忽略可能的原因。此外，在常用於單側偏癱腦性麻痺孩童的神經復健方案當中，孩童的發展性忽略只有在雙側密集訓練後有顯著降低，可能是因為雙側密集訓練不僅增進了患側手的能力，同時也提供了學習如何使用雙手的機會。未來可以根據本研究所提出的兩個觀點找出更多預測發展性忽略的重要因子，藉以設計更精準的治療方案。

Background and purposes Children with unilateral cerebral palsy (UCP) often tend not to use their more-affected hands, even though the capacities have remained. This phenomenon is known as developmental disregard (DD). To ameliorate the phenomenon of DD is one of the ultimate goals of rehabilitation for children with UCP. Thus, unraveling the phenomenon of DD has high clinical value for choosing or designing precise therapy. The study series aimed to investigate the phenomenon of DD in three domains: assessment, mechanisms, and interventions. This research consisted of three aims that were achieved in three studies. Study 1 aimed to investigate the validity and reliability of the revised version of the Observation Test of Capacity, Performance, and Developmental Disregard (OTCPDD-R). Study 2 aimed to find the underlying factors of the phenomenon of DD based on two perspectives: the more-affected hand not being good to use and not being familiar with how to use the more-affected hand. Underlying factors including sensorimotor functions, visuospatial attention, and familiarity were discussed. Study 3 aimed to investigate the efficacy of constraint-induced movement therapy (CIMT) and bilateral intensive training (BIT) on the phenomenon of DD. Methods In study 1, 26 children with UCP were recruited to estimate the construct validity and the intrarater and interrater reliabilities of the OTCPDD-R. There were three parts in study 2 for investigating underlying factors to explain DD. In part 1, 33 children with UCP were recruited. The Melbourne Assessment 2 (MA2), Semmes–Weinstein monofilaments (SWM), Two-point discrimination (TPD), Stereognosis and OTCPDD-R were performed. In part 2, 16 children with UCP and 17 typically developing children were recruited. Both groups performed two types of visual search tasks and OTCPDD-R. In part 3, 10 children with UCP and 12 typically developing children were recruited. The OTCPDD-R and EEG measurements during motor imagery were performed. In study 3, 32 children with UCP were assigned to CIMT and BIT day camps. The total amount of training for each program was 36 hours. The OTCPDD-R was assessed before and immediately after the intervention. Results The construct validity of the OTCPDD-R was confirmed. The intrarater and interrater reliabilities were good to excellent. Relationships were found between DD scores and basic motor abilities, tactile registration, and spatial tactile perception. Higher severity of DD was associated with lower performance of the factors stated above. For visuospatial attention, the children with UCP showed equivalent performance on the feature and conjunction search if motor impairments were considered. The DD scores were associated with d’ in feature search (size 16) and conjunction search (size 8 and size 16). For the familiarity with the bimanual tasks, the children with UCP were more familiar with using their more-affected hands than the typically developing children. Moreover, the association between laterality index (LI) of ERD and DD scores was only found in the condition of using both hands. Children with higher severity of DD demonstrated the higher values of LI as they imagined using both hands. For using the CIMT and BIT to reduce the phenomenon of DD, only BIT demonstrated significant improvement after the training. Conclusion The OTCPDD-R is a valid and reliable tool for measuring the phenomenon of DD in children with UCP. For explaining the phenomenon of DD, the factors based on the two perspectives (not being good to use and not being familiar with how to use the more-affected hand) were related to the DD scores. These factors could contribute to the design of optimal interventions for reversing the phenomenon of DD. Regarding the interventions, the BIT had superior improvements on the phenomenon of DD. This result could be explained by the fact that the BIT, which emphasized the bimanual coordination during the intervention, might not only improve the abilities of the more-affected hand but also provide opportunities to know how to use both hands during bimanual tasks.