Mazumdar Jayitri, Chowdhury Priyanka, Bhattacharya Tunisha, Mondal Badal Chandra, Ghosh Utpal
Senior Resident, Department of Paediatrics, Calcutta National Medical College and Hospital, Kolkata, West Bengal, India.
Resaerch Scholar, Department of Biochemistry and Biophysics, University of Kalyani, West Bengal, India.
J Clin Diagn Res. 2017 Aug;11(8):GR01-GR06. doi: 10.7860/JCDR/2017/26960.10516. Epub 2017 Aug 1.
Congenital limb anomalies are outcome of improper bone formation during embryonic development when cells divide, differentiate with high rate. So, telomerase activity is essential to maintain telomere length for such highly dividing cells. Here, we report four cases of congenital limb anomalies with detailed structures of limbs along with other clinical manifestations of age less than two years. We compared telomere length, expression of telomerase and telomere-associated genes of Peripheral Blood Mononuclear Cells (PBMC) in patient and four age-matched normal individual. Patient-1 was diagnosed with congenital limb hypogenesis ectrodactyly sequence, an autosomal dominant disorder, showing absence of digits and fibula in upper and lower limb respectively. Both mother and grandmother of Patient-1 showed similar hypogenesis of limbs. Patient-2 showed bilateral clenched hand with arthrogryposis, microcephaly and holoprosencephaly. Both Patient-3 and Patient-4 has no radius in upper limb. Additionally, Paient-3 showed right sided orbital Space Occupying Lesion (SOL) and Paranasal Sinuses (PNS) whereas Patient-4 showed fused kidney with fanconi anaemia. Furthermore, all the patients showed shorter telomere length, inactive telomerase and de-regulated expression of telomere-associated proteins in PBMC compared with age-matched control group. So, we can conclude that congenital limb anomalies may be linked with telomeropathy and a study with large number of samples is required to firmly establish such association.
先天性肢体异常是胚胎发育过程中细胞高速分裂和分化时骨骼形成不当的结果。因此,端粒酶活性对于维持此类高度分裂细胞的端粒长度至关重要。在此,我们报告了4例先天性肢体异常病例,详细描述了肢体结构以及其他年龄小于2岁的临床表现。我们比较了患者和4名年龄匹配的正常个体外周血单个核细胞(PBMC)的端粒长度、端粒酶表达和端粒相关基因。患者1被诊断为先天性肢体发育不全并指缺指序列征,一种常染色体显性疾病,分别表现为上肢和下肢的指骨和腓骨缺失。患者1的母亲和祖母均表现出类似的肢体发育不全。患者2表现为双侧握拳并有关节挛缩、小头畸形和前脑无裂畸形。患者3和患者4上肢均无桡骨。此外,患者3右侧眼眶有占位性病变(SOL)和鼻窦(PNS),而患者4表现为融合肾并伴有范科尼贫血。此外,与年龄匹配的对照组相比,所有患者的PBMC均表现出较短的端粒长度、无活性的端粒酶以及端粒相关蛋白的表达失调。因此,我们可以得出结论,先天性肢体异常可能与端粒病有关,需要进行大量样本研究以牢固确立这种关联。