臺灣博碩士論文加值系統

前言：唐氏症患者發展遲緩及諸多先天性的健康缺損，帶來的經濟負擔相當龐大。而現今篩檢策略有多種，其中包括最新的胎兒母血游離DNA篩檢，準確度比起現有的篩檢方式精確，但價格也昂貴許多。世界各國已開始探討如何選擇唐氏症篩檢策略，本研究則是欲探討台灣的唐氏症篩檢成本效果分析。
研究目的：本研究利用成本效果分析方法，評估台灣不同種的唐氏症篩檢策略。
研究方法：本研究樣本為台灣地區2016年孕婦人數共210,267人，35歲以上共57151人，34歲以下則為153,116人。研究設計以決策樹模型對35歲以上孕婦進行以下六種篩檢策略的成本效果分析: 羊膜穿刺術、第一孕期唐氏症篩檢（first trimester screening, FTS）、第二孕期唐氏症篩檢(second trimester screening, STS)、應變式第一孕期篩檢、應變式第二孕期篩檢及全面cfDNA篩檢；34歲以下孕婦則扣掉羊膜穿刺術，進行其他五種策略的成本效果分析。效果參數來自國外文獻查證，成本的參數則使用各地衛生局的醫療機構自費項目。另外考慮參數的不確定性，藉由蒙地卡羅模擬，進行機率性敏感度分析。結果呈現為不同篩檢策略下之增加成本效果比(incremental cost-effectiveness ratio, ICER)、成本效果接受曲線(acceptability curve)。最大願付價格(willingness to pay, WTP)參考文獻設定為新台幣一千萬元。
研究結果： 若WTP閾值為新台幣一千萬元，35歲以上孕婦以羊膜穿刺術比起成本最低的第二孕期唐氏症篩檢其增加成本效果比為690萬元新台幣；但若WTP閾值小於五百萬元，則以FTS相對於STS的增加成本效果比等於871,610為最低。而34歲以下孕婦若WTP閾值為新台幣一千萬元，FTS比起成本最低的STS其增加成本效果比為4,981,156元。至於cfDNA篩檢因價格過於昂貴，無法成為優勢策略。但若以醫源性流產的效果來看，應變式第二孕期唐氏症篩檢具成本效果。
結論：若WTP閾值為新台幣一千萬元，本研究結果顯示34歲以下孕婦接受第一孕期唐氏症篩檢、35歲以上孕婦接受羊膜穿刺，在診斷唐氏症的結果上是最具成本效果的策略，而全面cfDNA篩檢則不具有成本效果。但若是考量到醫源性流產的結果，則不論產婦的年齡為35歲上下，皆以包含cfDNA的應變式第二孕期唐氏症篩檢為優勢策略。

This study is a decision tree model analyzing 6 Down screening strategies in pregnant women older than 35 years old and 5 strategies in those younger than 34 years old in Taiwan. The 6 strategies include amniocentesis, first trimester screening, second trimester screening, contingent first trimester screening, contingent second trimester screening and cfDNA screening. The 5 strategies for pregnancies younger than 34 years old are the remaining except amniocentesis. The target population was all pregnant women of Taiwan in 2016. Comparing the 6 strategies for pregnancies above 35 years old, amniocentesis had an incremental cost-effectiveness ratio lower than maximum willingness-to-pay and had highest probability of being cost-effective in acceptability curve. In pregnancies under 34 years old, first trimester screening is the most cost-effective strategy. Universal cfDNA screening is hard to be cost-effective due to its high cost. Nevertheless, contingent second trimester screening might be most cost-effective in terms of the outcome of procedure related fetal loss.

目錄
第壹章 前言 1
第1節. 研究背景 1
第2節. 研究目的 2
第貳章 文獻回顧 4
第1節. 唐氏症簡介 4
第2節. 唐氏症的診斷及篩檢 5
第3節. 唐氏症篩檢的種類 5
第4節. 唐氏症的確診方式 7
第5節. 唐氏症篩檢的偵測率 8
第6節. 羊膜穿刺術的流產風險 10
第7節. 健康科技的經濟評估 10
7-1 常見的健康科技評估方法 10
第8節. 唐氏症篩檢的成本效果分析研究 12
第參章 研究方法 18
第1節. 研究問題 18
第2節. 臨床事件或決策的描述 18
第3節. 分析模型 19
第4節. 研究假設 23
第5節. 參數來源 23
5-1 事件機率 26
5-2 篩檢工具的敏感度及特異度 28
5-3 成本分析 28
5-4 效果分析 30
5-5 成本效果分析 30
第6節. 統計分析 31
6-1 分析軟體 31
6-2 基準值分析 31
6-3 成本效果平面圖 32
6-4 決定性(DETERMINISTIC)及隨機性(PROBABILISTIC)敏感度分析 32
6-5 成本效果平面散佈圖 36
6-6 成本效果接受曲線（COST-EFFECTIVENESS ACCEPTABILITY CURVE；CEAC） 36
6-7 淨貨幣效益(NET MONETARY BENEFIT) 37
第肆章 研究結果 38
第1節. 決定性模式之成本效果分析 38
第2節. 單維敏感度分析 51
2-1 34歲以下單維敏感度分析 52
2-2 35歲以上單維敏感度分析 54
第3節. 機率性模式成本效果分析 55
第伍章 討論 63
第1節. 主要發現 63
第2節. 與文獻上唐氏症篩檢決策樹模型文章比較 64
第3節. 對國家唐氏症篩檢政策之建議 65
第4節. 本研究方法學的優點 65
第5節. 研究限制 66
第陸章 結論 68
參考文獻 69


表目錄
表 1 母血胎兒游離DNA(cfDNA)篩檢準確性文獻 22
表 2 唐氏症篩檢成本效果分析文獻整理 26
表 3 模型中參數基準值及範圍 38
表 4 台灣2016年各年齡分組產婦總人數 40
表 5 成本費用基準值及上下限 42
表 6 機率敏感度分析參數分布表 47
表 7 34歲以下孕婦接受唐氏症篩檢的成本效果分析結果(唐氏症診斷個案數) 52
表 8 35歲以上孕婦接受唐氏症篩檢的成本效果分析結果(唐氏症診斷個案數) 55
表 9 34歲以下孕婦接受唐氏症篩檢的成本效果分析結果(醫源性流產個案數) 57
表 10 35歲以上孕婦接受唐氏症篩檢的成本效果分析結果(醫源性流產個案數) 59
表 11 34歲以下孕婦接受唐氏症篩檢的成本效果分析結果(唐氏症偽陰性個案數) 61
表 12 35歲以上孕婦接受唐氏症篩檢的成本效果分析結果(唐氏症偽陰性個案數) 63
表 13 34歲以下孕婦唐氏症篩檢的成本與減少支出 73
表 14 35歲以上孕婦唐氏症篩檢的成本與減少支出 74

圖目錄
圖 1 35歲以上唐氏症篩檢的決策樹模型 33
圖 2 34歲以下唐氏症篩檢的決策樹模型 34
圖 3 全面進行羊膜穿刺術之決策樹分枝 34
圖 4 全面進行初期唐氏症篩檢/中期唐氏症篩檢/cfDNA篩檢之決策樹分枝 35
圖 5 全面進行應變性初期唐氏症篩檢/應變性中期唐氏症篩檢之決策樹分枝 36
圖 6 53
圖 7 56
圖 8 58
圖 9. 60
圖 10 62
圖 11 64
圖 12 65
圖 13 66
圖 14 66
圖 15 67
圖 16 34歲以下機率性成本效果分佈圖 69
圖 17 34歲以下孕婦成本效果接受曲線 70
圖 18 35歲以上機率性成本效果分佈圖 71
圖 19 35歲以上孕婦成本效果接受曲線 72

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