Growth patterns in individuals with CDKL5 deficiency disorder

Dev Med Child Neurol. 2024 Apr;66(4):469-482. doi: 10.1111/dmcn.15777. Epub 2023 Oct 7.

Abstract

Aim: To compare growth in individuals with cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder with population norms and to investigate the effect of gastrostomy on growth.

Method: The longitudinal study included 353 individuals from the International CDKL5 Disorder Database with any anthropometric measurement in baseline and/or follow-up questionnaires. The British 1990 growth reference was used to determine the age- and sex-standardized z-score. Repeated cross-sectional data were fitted using a Gaussian linear regression model with generalized estimating equations.

Results: All growth parameters were below the general population norm (mean z-scores: weight -0.97, height -0.65, body mass index [BMI] -0.81, head circumference -2.12). The disparity was particularly pronounced for all anthropometric measurements after 4 years of age except for BMI. Moreover, individuals with gastrostomy placement were shown to have a larger decrease than those without.

Interpretation: In addition to weight, height, and BMI, head circumference was also compromised in this disorder. Microcephaly could be considered a helpful diagnostic feature, especially in adults. Any benefit of gastrostomy on weight and BMI was mainly seen in the early years.

MeSH terms

  • Adult
  • Body Height*
  • Body Mass Index
  • Body Weight
  • Cross-Sectional Studies
  • Epileptic Syndromes*
  • Humans
  • Longitudinal Studies
  • Protein Serine-Threonine Kinases* / genetics
  • Spasms, Infantile*

Substances

  • CDKL5 protein, human
  • Protein Serine-Threonine Kinases

Supplementary concepts

  • CDKL5 deficiency disorder