Barroso Sonia, Rieubland Claudine, José álvarez Antonia, López-Trascasa Margarita, Bart Pierre-Alexandre, Núñez-Roldán Antonio, Sánchez Berta
Servicio de Inmunología, Hospital Universitario Virgen del Rocío, Sevilla, Spain.
Immunology. 2006 Jun;118(2):257-60. doi: 10.1111/j.1365-2567.2006.02364.x.
Different genetic mutations have been described in complement components resulting in total or subtotal deficiency states. In this work we report the genetic basis of C7 deficiency in a previously reported Spanish patient exhibiting a combined total deficiency of C7 and C4B associated with systemic lupus erythematosus. Exon-specific polymerase chain reaction and sequencing revealed a not previously described single base mutation in exon 10 (T1458A) leading to a stop codon that causes the premature truncation of the C7 protein (C464X). Additionally, a C to A transversion at position 1561 (exon 11) was found in the patient resulting in an amino acid change (R499S). This latter mutation has been previously reported in individuals with subtotal C7 deficiency or with combined subtotal C6/C7 deficiency from widely spaced geographical areas. Another novel mutation was found in a second patient with meningococcal meningitis of Bolivian and Czech origin; a 11-base pair deletion of nucleotides 631-641 in exon 6 leading to the generation of a downstream stop codon causing the premature truncation of the C7 protein product (T189 x 193). This patient was found to be a heterozygous compound for another mutation in C7; a two-base pair deletion of nucleotides 1922 and 1923, 1923 and 1924 or 1924 and 1925 in exon 14 (1922delAG/1923delGA/1924delAG), leading again to the generation of a downstream stop codon that provokes the truncation of the C7 protein (S620x630). This latter mutation has been recently reported by our group in another Spanish family. Our results provide more evidences for the heterogeneous molecular basis of C7 deficiency.
已有文献报道补体成分存在不同的基因突变,可导致完全或部分缺陷状态。在本研究中,我们报告了一名先前报道的西班牙患者C7缺陷的遗传基础,该患者表现为C7和C4B完全缺乏,并与系统性红斑狼疮相关。外显子特异性聚合酶链反应和测序显示,外显子10存在一个先前未描述的单碱基突变(T1458A),导致产生一个终止密码子,致使C7蛋白过早截断(C464X)。此外,在该患者中还发现第1561位(外显子11)发生了C到A的颠换,导致氨基酸改变(R499S)。后一种突变先前已在来自广泛地理区域的C7部分缺乏或C6/C7联合部分缺乏的个体中报道过。在另一名患有玻利维亚和捷克血统的脑膜炎球菌性脑膜炎的患者中发现了另一个新突变;外显子6中核苷酸631 - 641缺失11个碱基对,导致产生一个下游终止密码子,致使C7蛋白产物过早截断(T189 x 193)。该患者被发现是C7另一个突变的杂合复合子;外显子14中核苷酸1922和1923、1923和1924或1924和1925缺失两个碱基对(1922delAG/1923delGA/1924delAG),再次导致产生一个下游终止密码子,引发C7蛋白截断(S620x630)。我们小组最近在另一个西班牙家族中报道了后一种突变。我们的结果为C7缺陷的异质分子基础提供了更多证据。