Targeted massively parallel sequencing for congenital generalized lipodystrophy

Arch Endocrinol Metab. 2021 May 18;64(5):559-566. doi: 10.20945/2359-3997000000278.

Abstract

Objective: Our aim is to establish genetic diagnosis of congenital generalized lipodystrophy (CGL) using targeted massively parallel sequencing (MPS), also known as next-generation sequencing (NGS).

Methods: Nine unrelated individuals with a clinical diagnosis of CGL were recruited. We used a customized panel to capture genes related to genetic lipodystrophies. DNA libraries were generated, sequenced using the Illumina MiSeq, and bioinformatics analysis was performed.

Results: An accurate genetic diagnosis was stated for all nine patients. Four had pathogenic variants in AGPAT2 and three in BSCL2. Three large homozygous deletions in AGPAT2 were identified by copy-number variant analysis.

Conclusion: Although we have found allelic variants in only 2 genes related to CGL, the panel was able to identify different variants including deletions that would have been missed by Sanger sequencing. We believe that MPS is a valuable tool for the genetic diagnosis of multi-genes related diseases, including CGL.

Keywords: Berardinelli-Seip syndrome; Congenital generalized lipodystrophy; deep sequencing; massively parallel sequencing.

MeSH terms

  • Alleles
  • GTP-Binding Protein gamma Subunits* / genetics
  • High-Throughput Nucleotide Sequencing
  • Humans
  • Lipodystrophy* / diagnosis
  • Lipodystrophy* / genetics
  • Lipodystrophy, Congenital Generalized* / diagnosis
  • Lipodystrophy, Congenital Generalized* / genetics
  • Mutation / genetics

Substances

  • BSCL2 protein, human
  • GTP-Binding Protein gamma Subunits