Rett syndrome diagnostic odyssey: Limitations of NextGen sequencing

Am J Med Genet A. 2024 Oct;194(10):e63725. doi: 10.1002/ajmg.a.63725. Epub 2024 May 22.

Abstract

Typical (or classic) Rett syndrome (RTT) is an X-linked neurodevelopmental disorder characterized by a period of regression, partial or complete loss of purposeful hand movements, and acquired speech, impaired gait, and stereotyped hand movements. In over 95% of typical RTT, a pathogenic variant is found in the methyl-CPG binding protein 2 gene (MECP2). Here, we describe a young woman with clinically diagnosed typical RTT syndrome who lacked a genetic diagnosis despite 20 years of investigation and multiple rounds of sequencing the MECP2 gene. Recently, additional genetic testing using next-generation sequencing was completed, which revealed a partial insertion of the BCL11A gene within exon 4 of MECP2, resulting in a small deletion in MECP2, causing likely disruption of MeCP2 function due to a frameshift. This case demonstrates the ever-changing limitations of genetic testing, as well as the importance of continual pursuit of a diagnosis as technologies improve and are more widely utilized.

Keywords: MECP2; Rett syndrome; next‐generation sequencing (NGS).

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Exons / genetics
  • Female
  • Frameshift Mutation / genetics
  • Genetic Testing*
  • High-Throughput Nucleotide Sequencing*
  • Humans
  • Methyl-CpG-Binding Protein 2* / genetics
  • Phenotype
  • Rett Syndrome* / diagnosis
  • Rett Syndrome* / genetics

Substances

  • Methyl-CpG-Binding Protein 2
  • MECP2 protein, human