Decline in gross motor skills in adult Rett syndrome; results from a Danish longitudinal study

Am J Med Genet A. 2021 Dec;185(12):3683-3693. doi: 10.1002/ajmg.a.62429. Epub 2021 Jul 23.

Abstract

Longevity of individuals with neurodevelopmental diseases as Rett syndrome (RTT) has increased and many reach adulthood and old age. There is therefore a need to increase knowledge about the course of RTT in adults in order to improve medical care management and quality of life. We did a longitudinal study to address if a possible decline in motor skills in adults with RTT can be explained by the presence of common medical conditions as epilepsy, breathing disturbance, and scoliosis. Data from the Danish RTT database, medical files, and videos from visits at the national Center for Rett syndrome were reviewed. The study included 24 individuals aged 30-66 years at last visit after a follow-up period of 6-12 years. Results showed a clinically observable and significant decline in gross motor skills using the Rett syndrome Gross Motor Scale (RSGMS) with a tendency of less decline in the individuals with the best motor abilities. The frequencies of comorbidities were high. Decline in RSGMS score was associated with the presence of epilepsy and severe scoliosis that had been conservatively managed. The results emphasize that epilepsy plays a significant role in the adult RTT life and management of severe scoliosis in the younger years has impact on the motor abilities in adulthood.

Keywords: Rett syndrome; adult; aging; longitudinal study; motor skills.

MeSH terms

  • Adult
  • Aged
  • Denmark
  • Epilepsy / complications
  • Epilepsy / physiopathology
  • Female
  • Follow-Up Studies
  • Humans
  • Longevity / genetics*
  • Longevity / physiology
  • Longitudinal Studies
  • Male
  • Middle Aged
  • Motor Skills / physiology
  • Motor Skills Disorders / complications
  • Motor Skills Disorders / epidemiology
  • Motor Skills Disorders / physiopathology*
  • Neurodevelopmental Disorders / complications
  • Neurodevelopmental Disorders / epidemiology
  • Neurodevelopmental Disorders / physiopathology*
  • Rett Syndrome / complications
  • Rett Syndrome / epidemiology
  • Rett Syndrome / physiopathology*
  • Severity of Illness Index