Duke University School of Medicine, Durham, NC, USA.
Department of Surgery, Duke University School of Medicine, Durham, NC, USA.
J Pediatr Urol. 2020 Jun;16(3):384.e1-384.e8. doi: 10.1016/j.jpurol.2020.03.023. Epub 2020 Apr 27.
Differences of sex development (DSD) engender ethical, social and psychosexual complexities that can complicate medical decision-making. We performed a web-based pilot study to estimate the utility value of a DSD diagnosis and to identify community concerns regarding DSD management.
A cross-sectional survey was posted on Amazon's Mechanical Turk, an online crowdsourcing platform. Respondents were ≥18y and were randomized to receive information on one of three common DSD conditions: Congenital Adrenal Hyperplasia (CAH), Mixed Gonadal Dysgenesis (MGD), and Partial Androgen Insensitivity Syndrome (PAIS). Time trade-off methodology was used to estimate utility values. Likert scale and statement-ranking questions were used to assess respondent perceptions.
Of 1,628 respondents, median age was 34y; most respondents were parents (59.1%), white (77.1%), and previously unfamiliar with DSD (60.4%). The median overall utility value was 0.70 (IQR 0.50-0.90), similar to moderately severe chronic health conditions. Utility estimates varied based on the DSD scenario presented (0.80 CAH vs. 0.70 MGD vs. 0.80 PAIS, p = 0.0006), respondent gender (p < 0.0001), race (p = 0.002), religion (p = 0.005), and prior knowledge of DSD (p < 0.0001). Reported concerns included gender identity (23.4%), urinary function (20.5%) and surgical complications (17.4%). Most (67.5%) supported early surgical intervention at 6-18 mo; 10.4% thought surgery should occur ≥18 y.
Limitations of this study include that survey participants were aware of the nature of the study, thus some respondents may have participated to skew the results. Given the nature of this pilot study, the representation of families with children with DSD within the study is severely limited given the rarity of DSDs. This means that their opinions may be diluted by the large sample size. However, because utility values are classically estimated according to community opinions, the utility data presented should be taken to reflect that of the specific sample studied and is not reflective of that of families with a vested interest in such cases.
Community-based respondents perceived that DSD conditions were associated with a reduction in utility values (0.70-0.80), on par with moderately severe chronic health conditions. Estimates varied based on respondents' gender, race, religion and prior knowledge of DSD. Gender identity was the most concerning aspect for respondents.
性别发育差异（DSD）引发了伦理、社会和心理性别方面的复杂性，这可能使医疗决策复杂化。我们进行了一项基于网络的试点研究，以评估 DSD 诊断的实用价值，并确定社区对 DSD 管理的关注。
横断面调查发布在亚马逊的 Mechanical Turk 上，这是一个在线众包平台。受访者年龄≥18 岁，并随机分为三组，分别接受三种常见 DSD 情况的信息：先天性肾上腺增生症（CAH）、混合性性腺发育不良（MGD）和部分雄激素不敏感综合征（PAIS）。时间权衡方法用于估计实用价值。李克特量表和陈述排名问题用于评估受访者的看法。
在 1628 名受访者中，中位年龄为 34 岁；大多数受访者是父母（59.1%），白人（77.1%），以前不了解 DSD（60.4%）。总体实用价值的中位数为 0.70（IQR 0.50-0.90），与中度严重的慢性健康状况相当。效用估计值因呈现的 DSD 情况而异（0.80 CAH 与 0.70 MGD 与 0.80 PAIS，p=0.0006），受访者性别（p<0.0001）、种族（p=0.002）、宗教（p=0.005）和对 DSD 的先验知识（p<0.0001）。报告的担忧包括性别认同（23.4%）、尿功能（20.5%）和手术并发症（17.4%）。大多数人（67.5%）支持在 6-18 个月时进行早期手术干预；10.4%的人认为手术应该在 18 岁以上进行。
本研究的局限性包括调查参与者了解研究的性质，因此一些参与者可能参与其中以歪曲结果。鉴于本试点研究的性质，由于 DSD 的罕见性，研究中患有 DSD 儿童的家庭的代表性严重受限。这意味着他们的意见可能因样本量大而被稀释。然而，由于实用价值通常根据社区意见进行估计，因此提出的实用价值数据应反映特定样本的意见，而不是对有兴趣参与此类案例的家庭的意见。
基于社区的受访者认为 DSD 状况与实用价值降低（0.70-0.80）相关，与中度严重的慢性健康状况相当。估计值因受访者的性别、种族、宗教信仰和对 DSD 的先验知识而异。性别认同是受访者最关心的方面。
