Department of Hematology, Christian Medical College, Vellore, Tamil Nadu, India.
Haemophilia. 2012 May;18(3):e195-200. doi: 10.1111/j.1365-2516.2011.02730.x. Epub 2012 Jan 4.
Data on the clinical manifestations of patients with clotting factor defects other than Haemophilia A, B and von Willebrand disease are limited because of their rarity. Due to their autosomal recessive nature of inheritance, these diseases are more common in areas where there is higher prevalence of consanguinity. There is no previous large series reported from southern India where consanguinity is common. Our aim was to analyze clinical manifestations of patients with rare bleeding disorders and correlate their bleeding symptoms with corresponding factor level. Data were collected in a standardized format from our centre over three decades on 281 patients who were diagnosed with rare bleeding disorders (fibrinogen, prothrombin, factor V (FV), FVII, FX, FXI, FXIII and combined FV or FVIII deficiency). Patients with liver dysfunction or those on medications which can affect factor level were excluded. All patients with <50% factor levels were included in this analysis. Patients were analysed for their salient clinical manifestations and it was correlated with their factor levels. The data shows that FXIII deficiency is the commonest and FXI deficiency is the rarest in Southern India. There was no significant difference in bleeding symptoms among those who were < or >1% factor coagulant activities among all disorders, except for few symptoms in FVII and FX deficiency. An international collaborative study is essential to find out the best way of classifying severity in patients with rare bleeding disorders.
有关除血友病 A、B 和血管性血友病以外的凝血因子缺陷患者的临床表现的数据十分有限,这是因为此类疾病较为罕见。由于这些疾病的遗传方式为常染色体隐性遗传,因此在近亲结婚率较高的地区更为常见。在近亲结婚较为常见的印度南部,尚无此前对此类罕见出血性疾病患者进行的大型系列报道。我们旨在分析患有罕见出血性疾病患者的临床表现,并将其出血症状与相应的因子水平相关联。我们在 30 多年的时间里,以标准化格式从我们的中心收集了 281 名患有罕见出血性疾病(纤维蛋白原、凝血酶原、因子 V(FV)、因子 VII(FVII)、因子 X(FX)、因子 XI(FXI)、因子 XIII 和 FV 或 FVIII 联合缺乏)患者的数据。排除了有肝功能障碍或正在服用可能影响因子水平的药物的患者。所有因子水平<50%的患者均纳入本分析。对患者的显著临床表现进行分析,并与他们的因子水平相关联。数据显示,在印度南部,FXIII 缺乏症最为常见,FXI 缺乏症最为罕见。除 FVII 和 FX 缺乏症的少数症状外,在所有疾病中,<或>1%因子凝血活性的患者之间的出血症状并无显著差异。开展国际合作研究对于找到对罕见出血性疾病患者进行严重程度分类的最佳方法至关重要。