Department of Medicine, University of Alberta, Edmonton, Canada.
Mazankowski Alberta Heart Institute, University of Alberta, Edmonton, Canada.
PLoS One. 2020 Sep 28;15(9):e0239675. doi: 10.1371/journal.pone.0239675. eCollection 2020.
Fabry Disease (FD) is a systemic disorder that can result in cardiovascular, renal, and neurovascular disease leading to reduced life expectancy. FD should be considered in the differential of all patients with unexplained left ventricular hypertrophy (LVH). We therefore performed a prospective screening study in Edmonton and Hong Kong using Dried Blood Spot (DBS) testing on patients with undiagnosed LVH. Participants found to have unexplained LVH on echocardiography were invited to participate and subsequently subjected to DBS testing. DBS testing was used to measure α-galactosidase (α-GAL) enzyme activity and for mutation analysis of the α-galactosidase (GLA) gene, both of which are required to make a diagnosis of FD. DBS testing was performed as a screening tool on patients (n = 266) in Edmonton and Hong Kong, allowing for detection of five patients with FD (2% prevalence of FD) and one patient with hydroxychloroquine-induced phenocopy. Left ventricular mass index (LVMI) by GLA genotype showed a higher LVMI in patients with IVS4 + 919G > A mutations compared to those without the mutation. Two patients were initiated on ERT and hydroxychloroquine was discontinued in the patient with a phenocopy of FD. Overall, we detected FD in 2% of our screening cohort using DBS testing as an effective and easy to administer screening tool in patients with unexplained LVH. Utilizing DBS testing to screen for FD in patients with otherwise undiagnosed LVH is clinically important due to the availability of effective therapies and the value of cascade screening in extended families.
法布里病(FD)是一种全身性疾病,可导致心血管、肾脏和神经血管疾病,从而降低预期寿命。在所有原因不明的左心室肥厚(LVH)患者中,都应考虑 FD 的鉴别诊断。因此,我们在埃德蒙顿和香港进行了一项前瞻性筛查研究,对未经诊断的 LVH 患者使用干血斑(DBS)检测。通过超声心动图发现存在原因不明的 LVH 的参与者被邀请参加,并随后接受 DBS 检测。DBS 检测用于测量α-半乳糖苷酶(α-GAL)酶活性和α-半乳糖苷酶(GLA)基因的突变分析,这两者都是 FD 诊断所必需的。在埃德蒙顿和香港的患者中,DBS 检测作为一种筛查工具进行(n=266),可以检测到 5 例 FD 患者(FD 的患病率为 2%)和 1 例羟氯喹诱导的表型患者。根据 GLA 基因型的左心室质量指数(LVMI)显示,与没有突变的患者相比,IVS4+919G>A 突变患者的 LVMI 更高。两名患者开始接受 ERT,FD 表型患者停用了羟氯喹。总的来说,我们使用 DBS 检测在 2%的筛查队列中检测到 FD,这是一种有效且易于管理的筛查工具,用于原因不明的 LVH 患者。由于有效的治疗方法和在扩展家族中进行级联筛查的价值,利用 DBS 检测对其他未诊断的 LVH 患者进行 FD 筛查在临床上非常重要。