• Fulltext

       

        Click here to view fulltext PDF


      Permanent link:
      https://www.ias.ac.in/article/fulltext/jgen/101/0030

    • Keywords

       

      developmental encephalopathy; agenesis of corpus callosum; epilepsy; C12orf57; compound heterozygous.

    • Abstract

       

      Open reading frame variants which lack stop codons such as C12orf57 variants are known to cause Temtamy syndrome, an extremely rare disorder characterized by intellectual disability, seizures, facial dysmorphism and agenesis of corpus callosum. C12orf57 was initially reported to be required for human corpus callosum development. We report the first child who is of Indian origin with developmental and epileptic encephalopathy (DEE) with a unique phenotypic evolution as focal onset reflex seizures. We performed wholeexome sequencing of genomic DNA isolated from peripheral blood samples of proband and his parents. Two pathogenic compound heterozygous variants, a start loss variant (Chr12:7053285:c.1A>G) and a premature stop gain variant (Chr12:7053327:c.43C>T), involving the C12orf57 gene were identified in the proband. Our case report which details genotyping in this rare syndromic developmental encephalopathy, with no prior cases reported from India, expands the ethnic spectrum of patients.

    • Author Affiliations

       

      F. ALFIYA1 MANNA JOSE1 SOUMYA V. CHANDRASEKHARAN1 SOUMYA SUNDARAM1 MADHUSOODANAN URULANGODI2 BEJOY THOMAS3 ASHALATHA RADHAKRISHNAN1 MOINAK BANERJEE4 RAMSHEKHAR N. MENON1

      1. Department of Neurology, Sree Chitra Tirunal Institute for Medical Sciences and Technology (SCTIMST), Thiruvananthapuram 695 011, India
      2. Department of Biochemistry, Sree Chitra Tirunal Institute for Medical Sciences and Technology (SCTIMST), Thiruvananthapuram 695 011, India
      3. Department of Imaging Sciences, Sree Chitra Tirunal Institute for Medical Sciences and Technology (SCTIMST), Thiruvananthapuram 695 011, India
      4. Human Molecular Genetics Laboratory, Rajiv Gandhi Centre for Biotechnology, Thiruvananthapuram 695 014, India
    • Dates

       
  • Journal of Genetics | News

    • Editorial Note on Continuous Article Publication

      Posted on July 25, 2019

      Click here for Editorial Note on CAP Mode

© 2022-2023 Indian Academy of Sciences, Bengaluru.