Dabboubi Rym, Amri Yessine, Yahyaoui Salem, Mahjoub Rahma, Sahli Chayma Abdelhafidh, Sahli Chaima, Hadj Fredj Sondess, Bibi Amina, Sammoud Azza, Messaoud Taieb
Biochemistry Laboratory, Bechir Hamza Children's Hospital, Bab Saadoun Square, 1007, Tunis, Tunisia.
Biochemistry Laboratory, Bechir Hamza Children's Hospital, Bab Saadoun Square, 1007, Tunis, Tunisia.
Eur J Med Genet. 2020 May;63(5):103874. doi: 10.1016/j.ejmg.2020.103874. Epub 2020 Feb 3.
Congenital atransferrinemia is an extremely rare autosomal recessive disorder resulting in the complete absence or extremely reduced amount of transferrin. In this study, we describe the first case of congenital atransferrinemia in Tunisia and the 18th patient in the reported data. The patient was referred to our hospital to explore a severe hypochromic and microcytic anemia. The laboratory evaluation including hematological and biochemical examination was performed in the proband and her parents. All exons of the transferrin gene were PCR amplified. The products were screened for mutations by direct sequencing. Based on laboratory and clinical findings, diagnosis of congenital atransferrinemia was confirmed. DNA sequencing revealed the presence of a novel homozygous deletion (c.293-63del) in the intron 13. This mutation is predicted to generate a higher score cryptic branch point leading to the production of an altered mRNA molecule. The second previously reported missense mutation p.Arg609Trp. Crystallographic structure analyzes demonstrate that the mutation would probably lead to significant conformational change not allowing the expression of transferrin protein. Current molecular characterization of this novel transferrin abnormality puts to the proof the variability in onset, first blood transfusion, and phenotypic expression in atransferrinemic patients.
先天性无转铁蛋白血症是一种极为罕见的常染色体隐性疾病,会导致转铁蛋白完全缺失或含量极低。在本研究中,我们描述了突尼斯首例先天性无转铁蛋白血症病例,也是报告数据中的第18例患者。该患者因严重低色素小细胞性贫血前来我院就诊。对先证者及其父母进行了包括血液学和生化检查在内的实验室评估。转铁蛋白基因的所有外显子均通过PCR扩增。通过直接测序对产物进行突变筛查。基于实验室和临床检查结果,确诊为先天性无转铁蛋白血症。DNA测序显示内含子13存在一个新的纯合缺失(c.293 - 63del)。该突变预计会产生一个更高评分的隐蔽分支点,导致产生一个改变的mRNA分子。第二个是先前报道的错义突变p.Arg609Trp。晶体结构分析表明,该突变可能会导致显著的构象变化,从而使转铁蛋白无法表达。这种新的转铁蛋白异常的当前分子特征证明了无转铁蛋白血症患者在发病、首次输血和表型表达方面的变异性。