Department of Hematology, Peking Union Medical College, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China.
Department of Hematology, Peking Union Medical College, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China.
Leuk Res. 2022 Sep;120:106917. doi: 10.1016/j.leukres.2022.106917. Epub 2022 Jul 5.
Factor X (FX) deficiency is prevalent in light-chain (AL) amyloidosis but its clinical significance was not investigated deeply. We conducted a retrospective analysis of a consecutive cohort with 207 primary AL amyloidosis patients. FX deficiency was present in 129 patients (62.3%). Those with FX deficiency had higher dFLC (299.6 mg/L vs. 102.3 mg/L, P < 0.001), higher cardiac troponin I (0.05 μg/L vs. 0.02 μg/L, P < 0.001) and N-terminal pro-brain natriuretic peptide (3115 ng/L vs. 392 ng/L, P < 0.001), and more patients with bone marrow plasma cells > 10% (18.0% vs. 4.3%, P = 0.008). The prevalence of FX deficiency increased with the Mayo 2004 stage. FX-deficient patients exhibited inferior overall survival (P < 0.001) and progression-free survival (P < 0.001) than others. Fifty-five patients with FX deficiency received retesting of FX activity after anti-plasma cell therapy. The median variation in FX activity was + 6.8% (range, -24.5% ~ +73.4%). Better improvement of FX activity was observed in patients with complete hematologic response (+18.2% vs. +4.0%, P = 0.036) and at least one organ response (+14.4% vs. +3.4%, P = 0.024). FX deficiency is associated with a heavier disease burden and poorer survival in primary AL amyloidosis. Improvement of FX activity tends to appear in patients with better hematologic and organ responses after chemotherapy.
FX 因子缺乏症在轻链(AL)淀粉样变性中较为常见,但尚未深入研究其临床意义。我们对 207 例原发性 AL 淀粉样变性患者进行了回顾性队列分析。129 例(62.3%)存在 FX 因子缺乏症。FX 因子缺乏症患者的游离轻链(dFLC)更高(299.6mg/L 比 102.3mg/L,P<0.001),心脏肌钙蛋白 I(0.05μg/L 比 0.02μg/L,P<0.001)和 N 末端脑钠肽前体(3115ng/L 比 392ng/L,P<0.001)更高,骨髓浆细胞>10%的患者更多(18.0%比 4.3%,P=0.008)。随着 Mayo 2004 分期,FX 因子缺乏症的患病率增加。FX 缺乏的患者总生存(P<0.001)和无进展生存(P<0.001)均较差。55 例 FX 因子缺乏症患者在接受浆细胞靶向治疗后接受 FX 因子活性的重复检测。FX 因子活性的中位数变化为+6.8%(范围,-24.5%~+73.4%)。在完全血液学缓解(+18.2%比+4.0%,P=0.036)和至少一个器官缓解(+14.4%比+3.4%,P=0.024)的患者中,观察到 FX 因子活性的改善更为明显。FX 因子缺乏症与原发性 AL 淀粉样变性患者的疾病负担更重和生存较差相关。在化疗后血液学和器官反应更好的患者中,FX 因子活性的改善趋势更为明显。