Witt Stefanie, Rohenkohl Anja, Bullinger Monika, Sommer Rachel, Kahrs Sabine, Klingebiel Karl-Heinz, Klingebiel Ruzena, Quitmann Julia
University Medical Center Hamburg-Eppendorf, Department of Medical Psychology, Martinistr. 52, 20246 Hamburg, Germany.
German Association for People of Short Stature and their Families (BKMF e.V), Leinestrabe 2, 28199 Bremen, Germany.
Pediatr Endocrinol Rev. 2017 Nov;15(Suppl 1):109-118. doi: 10.17458/per.vol15.2017.wrm.improvinghealthrelatedquality.
Achondroplasia (ACH) is a rare, genetically determined health condition. Patients suffer from disproportional short stature and multiple physical and functional impairments as well as socioemotional problems. Despite the burden of disease, only few studies focus on health-related quality of life (HrQoL) of young ACH patients. In a series of studies, the BKMF e.V. in cooperation with the UKE studied ACH patients' and parents' experience of HrQoL, their responses to HrQoL questionnaires and their evaluation of a HrQoL based intervention. Both qualitative and quantitative approaches were used. Psychometrically appropriate instruments were identified and the wellbeing of young patients with ACH was analyzed showing no difference from a healthy norm sample using generic instruments. However, disease-specific instruments showed discrepancies between patients with proportional and disproportional short stature. Still, results show a significant effect by age and the evaluation of the counselling concept reveals that young ACH-patients especially benefit from such intervention.
软骨发育不全(ACH)是一种罕见的、由基因决定的健康状况。患者身材比例失调、身材矮小,伴有多种身体和功能障碍以及社会情感问题。尽管疾病负担沉重,但只有少数研究关注年轻ACH患者的健康相关生活质量(HrQoL)。在一系列研究中,德国软骨发育不全患者援助协会(BKMF e.V.)与汉堡大学艾本德儿童医院(UKE)合作,研究了ACH患者及其父母对HrQoL的体验、他们对HrQoL问卷的回答以及他们对基于HrQoL的干预措施的评估。研究采用了定性和定量两种方法。确定了心理测量学上合适的工具,并分析了ACH年轻患者的幸福感,结果显示使用通用工具时与健康标准样本没有差异。然而,疾病特异性工具显示身材比例正常和身材比例失调的患者之间存在差异。尽管如此,结果显示年龄有显著影响,对咨询概念的评估表明,年轻的ACH患者尤其受益于这种干预。