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  • 學位論文

龐貝氏症新生兒篩檢偽陽性事件對父母擔任親職之長期影響

Long-term effect of false-positive Pompe disease newborn screening on parenting

指導教授 : 吳英璋
共同指導教授 : 胡務亮(Wuh-Liang Hwu)

摘要


新生兒篩檢是一種 「以區域性人口族群」為背景的公共衛生政策,目的是降低某些先天代謝異常疾病的殘障率、疾病嚴重度或是死亡率。近年來,由於人類基因圖譜製作完成,衍生了許多新興科技、技術的發展與應用,也因此帶動了新生兒篩檢的發展,陸陸續續增加了新的疾病項目,龐貝氏症便是其中之一。本研究的目的是探討在某大醫院遺傳諮詢中心歷經龐貝氏症新生兒篩檢複檢及疾病確認事件後,篩檢結果為偽陽性之個案父母,其長期擔任親職時是否受到該複檢及疾病確認事件之影響。一共收集了研究組「有歷經龐貝氏症篩檢偽陽性的事件」之家長所填具的有效問卷19份,對照組「沒有歷經龐貝氏症篩檢偽陽性的事件」之家長所填具的有效問卷32份。 研究的結果發現:研究組與對照組在「疾病不確定感」、「親職壓力」、「親職勝任感」三大主變項總分的比較中,在「疾病不確定感」(P=0.04)主變項達到顯著差異,對照組得分較高,表示研究組的疾病不確定感高於對照組。在「缺乏資訊」、「不明確」、「模糊不清」、「無法預測」、「親職愁苦」、「互動失調」、「困難兒童」、「效能」、「挫折」九項次級變項的比較中,在「缺乏資訊」(P=0.003)與「不明確」(P=0.006)次級變項達到顯著差異,且對照組得分高於研究組,顯示研究組在「缺乏資訊」與「不明確」向度上的疾病不確定感比對照組來的高。另外,將研究組分為「擔心」與「不擔心」篩檢結果2組,再作比較,結果發現「擔心」與「不擔心」在三大主變項的比較中,均未達顯著差異,但是在「親職壓力」主變項中有達到顯著差異的趨勢(P=0.055);九項次級變項的比較中,則是在「互動失調」(P=0.008)次級變項中達到顯著差異,且在「親職愁苦」(P=0.057)次級變項中,有達到顯著差異的趨勢,且都是「不擔心」得分較高。此結果顯示「有歷經龐貝氏症篩檢偽陽性的事件」且在篩檢複檢與疾病確認過程中表達「擔心」的父母,在「互動失調」向度上的親職壓力,會比表達「不擔心」的父母高。 將研究組與對照組之間在基本資料上達顯著差異的「家庭月收入」與「孩童年齡」項目,以Pearson’s Correlation分析所有的樣本,檢驗這2個項目與九項次級變項得分以及三大變項得分之間的的相關性;結果發現「孩童年齡」與九項次級變項之間皆未達顯著相關,只有與「缺乏資訊」(相關係數=-0.284)次級變項得分之間有達到顯著負相關的趨勢。「家庭月收入」則「困難兒童」(相關係數=+0.292)次級變項得分呈現顯著正相關,表示在本研究樣本中,「家庭月收入」越高,「困難兒童」次級變項的得分也會越高,也就是在「困難兒童」向度的親職壓力會越低。三大變項之間皆呈現顯著正相關,相關係數分列如下: *「疾病不確定感」與「親職壓力」之間相關係數為+0.456,「疾病不確定感」得分越高,「親職壓力」得分也越高,也就是不確定感越低,親職壓力越低。 *「疾病不確定感」與「親職勝任感」之間相關係數為+0.519,「疾病不確定感」得分越高,「親職勝任感」得分也越高,也就是不確定感越低,親職勝任感越高。 *「親職壓力」與「親職勝任感」之間相關係數為+0.729,「親職壓力」得分越高,「親職勝任感」得分也越高,也就是親職壓力越低,親職勝任感越高。 顯示在本研究樣本中以「親職壓力」與「親職勝任感」之間的相關性最強,「疾病不確定感」與「親職勝任感」之間的相關性次之,「疾病不確定感」與「親職壓力」之間的相關性最弱。本研究中發現龐貝氏症新生兒篩檢偽陽性結果,長期來說並不會造成家長擔任親職時的親職壓力上升,但由於研究組與對照組在「疾病不確定感」主變項的比較中達到顯著差異,且本研究中也發現「疾病不確定感」、「親職壓力」、「親職勝任感」的得分有顯著正相關的關係;若是存有過高的不確定感,可能會造成親職勝任感降低與親職壓力變高。 這些歷經篩檢偽陽性事件的家長們要面對的是孩子可能患有重大疾病的疑慮,以及後續是否進行基因檢測的選擇,因此醫療照護體系中的訊息傳達與關係建立可能還需要再特別加以關注,所以提供相關的遺傳諮詢服務是有必要的。

並列摘要


Newborn screening is a public health policy aims at reducing disability, severity of illness, or mortality of inborn errors of metabolism. After the success of the human genome project a few years ago, new science and technology allow more diseases to be screened at the newborn stage, like Pompe disease. The purpose of this study is to explore long-term effect of Pompe disease newborn screening on parenting. We conducted 19 questionnaires on the parents whose babies have experienced a false-positive Pompe disease screening (the study group). We also conducted 32 questionnaires on parents whose babies have a negative Pompe disease screening (the control group). We found that the total scores of "Parenting Stress", and "Parenting Sense of Competence" are not significantly different between the study group and the control group. There is significantly different between the study group and the control group in the total score of "Uncertainty of Illness". Among the nine sub-variables ("Lack of Information" , " Lack of Clarity", "Multiattributed Ambiguity", "Unpredictability", "Parental distress", " Parent-Child dysfunctional interaction", "Difficult Child", "Efficiency", and "Frustration"), we found that the scores of "Lack of Information" and " Lack of Clarity" sub-variable are higher in the control group than in the study group. This suggests that the study group is more uncertain about illness than the control group. This result is not related to whether the parents are "worried" or "not worried" about the screening result. But these parents, who expressed "worried" about the screening result during false positive event, are perceiving more parenting stress in " Parent-Child dysfunctional interaction" dimension than expressed "not worried" about the screening result. One problem of the study is that the study group and the control group difference in both "family income" and "child age". We analyze these two items in respective to either the three main variables or the nine sub-variables by Pearson's Correlation. We find that "Family income" is positive correlated with "Difficult Child". Among the three main variables, each one is significantly positively correlated to the other two variables, respectively. In conclusion, a false positive Pompe disease newborn screening does not affect parenting significantly in "Parenting Stress" and "Parenting Sense of Competence", but the significantly different in "Lack of Clarity" needs an attention. We need to be careful about delivering messages of newborn screening. Test instruction is very important, because the parents may face the fact having their children suffered from major illness. Service of genetic counseling is certainly needed.

參考文獻


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