van Bergen Eline D P, Mastbergen Simon C, Lafeber Floris P J G, Bay-Jensen Anne-Christine, Madsen Sofie F, Port Helena, Foppen Wouter, Schutgens Roger E G, Jansen Mylène P, van Vulpen Lize F D
Center for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek, Heidelberglaan, The Netherlands.
Department of Rheumatology & Clinical Immunology, Center for Benign Haematology, Thrombosis and Haemostasis, Van Creveldkliniek, Heidelberglaan, The Netherlands.
Haemophilia. 2023 May;29(3):874-882. doi: 10.1111/hae.14784. Epub 2023 Apr 5.
Ankle joint distraction (AJD) is a promising treatment for patients with severe haemophilic ankle arthropathy (HAA). However, some patients showed no clinical improvement after AJD and these differences may be related to structural differences.
Primarily to quantify the structural changes after AJD in patients with HAA by the use of 3D joint space width (JSW) measurements and biochemical markers and secondarily to correlate these findings with clinical pain/function.
Patients with haemophilia A/B who underwent AJD were included for this study. Bone contours on MRI (performed before and 12 and 36 months after AJD) were drawn manually and percentage change in JSW was calculated. Blood/urine (before and 6, 12, 24 and 36 months after AJD) was collected for biomarker measurement (COMP, CS846, C10C, CALC2, PRO-C2, CTX-II) and combined indexes of markers were calculated. Mixed effects models were used for analyses on group level. Structural changes were compared with clinical parameters.
Eight patients were evaluated. On group level, percentage changes in JSW showed a slight decrease after 12 months followed by a non-statistically significant increase in JSW after 36 months compared to baseline. Biochemical marker collagen/cartilage formation also showed an initial decrease, followed by a trend towards net formation 12, 24 and 36 months after AJD. On individual patient level, no clear correlations between structural changes and clinical parameters were observed.
Cartilage restoration activity on group level in patients with HAA after AJD was in concordance with clinical improvements. Correlating structural modifications with clinical parameters in the individual patient remains difficult.
踝关节牵张术(AJD)是治疗重度血友病性踝关节病(HAA)患者的一种有前景的治疗方法。然而,一些患者在接受AJD治疗后并无临床改善,这些差异可能与结构差异有关。
主要通过三维关节间隙宽度(JSW)测量和生化标志物来量化HAA患者接受AJD治疗后的结构变化,其次将这些结果与临床疼痛/功能进行关联。
纳入接受AJD治疗的甲型/乙型血友病患者进行本研究。手动绘制MRI(AJD治疗前、治疗后12个月和36个月)上的骨轮廓,并计算JSW的百分比变化。收集血液/尿液(AJD治疗前、治疗后6个月、12个月、24个月和36个月)用于生物标志物测量(COMP、CS846、C10C、CALC2、PRO-C2、CTX-II),并计算标志物的综合指标。使用混合效应模型进行组水平分析。将结构变化与临床参数进行比较。
评估了8例患者。在组水平上,与基线相比,JSW百分比变化在12个月后略有下降,随后在36个月后JSW有非统计学意义的增加。生化标志物胶原蛋白/软骨形成也显示出最初的下降,随后在AJD治疗后12个月、24个月和36个月有净形成的趋势。在个体患者水平上,未观察到结构变化与临床参数之间的明确相关性。
HAA患者接受AJD治疗后,组水平上的软骨修复活性与临床改善一致。在个体患者中将结构改变与临床参数相关联仍然困难。