Severe presentation and complex brain malformations in an individual carrying a CCND2 variant

Mol Genet Genomic Med. 2019 Jun;7(6):e708. doi: 10.1002/mgg3.708. Epub 2019 May 6.

Abstract

Background: Megalencephaly-polymicrogyria-polydactyly-hydrocephalus (MPPH) is a developmental brain disorder characterized by megalencephaly and bilateral perisylvian polymicrogyria due to defects in genes of the PI3K-AKT pathway. Only a few patients with CCND2 mutations have been reported to date.

Methods: We describe an individual harboring a de novo variant in CCND2 undergoing neuroradiological evaluation including diffusion tensor imaging (DTI).

Results: The individual presented with a severe brain malformation extending to both brainstem and cerebellum with hypomyelination not previously reported in CCND2-related disorder. Severe hypoplasia and white matter disorganization were confirmed by DTI.

Conclusion: This report expands the phenotypic spectrum of the disorder due to CCND2 variants.

Keywords: CCND2; MPPH; Megalencephaly-polymicrogyria-polydactyly-hydrocephalus.

Publication types

  • Case Reports

MeSH terms

  • Cyclin D2 / genetics*
  • Diffusion Tensor Imaging
  • Humans
  • Hydrocephalus / diagnostic imaging
  • Hydrocephalus / genetics*
  • Hydrocephalus / pathology
  • Male
  • Malformations of Cortical Development / diagnostic imaging
  • Malformations of Cortical Development / genetics*
  • Malformations of Cortical Development / pathology
  • Mutation
  • Phenotype*
  • Polydactyly / diagnostic imaging
  • Polydactyly / genetics*
  • Polydactyly / pathology
  • Young Adult

Substances

  • CCND2 protein, human
  • Cyclin D2

Supplementary concepts

  • Megalancephaly Polymicrogyria-Polydactyly Hydrocephalus Syndrome