Literature DB >> 28969156

Patients with Congenital Limb Anomaly Show Short Telomere, Shutdown of Telomerase and Deregulated Expression of Various Telomere-Associated Proteins in Peripheral Blood Mononuclear Cells-A Case Series.

Jayitri Mazumdar1, Priyanka Chowdhury2, Tunisha Bhattacharya3, Badal Chandra Mondal4, Utpal Ghosh5.   

Abstract

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.

Entities:  

Keywords:  Embryonic developement; Shelterin protein; Telomere length; Telomeropathy; hTERC

Year:  2017        PMID: 28969156      PMCID: PMC5620797          DOI: 10.7860/JCDR/2017/26960.10516

Source DB:  PubMed          Journal:  J Clin Diagn Res        ISSN: 0973-709X


  34 in total

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Authors:  S A Kamranvar; X Chen; M G Masucci
Journal:  Oncogene       Date:  2013-05-27       Impact factor: 9.867

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