Login / Signup

CLN8 Mutations Presenting with a Phenotypic Continuum of Neuronal Ceroid Lipofuscinosis-Literature Review and Case Report.

Magdalena Badura-StronkaAnna Winczewska-WiktorAnna PietrzakAdam Sebastian HirschfeldTomasz ZemojtelKatarzyna WołyńskaKatarzyna Bednarek-RajewskaMonika Seget-DubaniewiczAgnieszka MatheiselAnna Latos-BielenskaBarbara Steinborn
Published in: Genes (2021)
CLN8 is a ubiquitously expressed membrane-spanning protein that localizes primarily in the ER, with partial localization in the ER-Golgi intermediate compartment. Mutations in CLN8 cause late-infantile neuronal ceroid lipofuscinosis (LINCL). We describe a female pediatric patient with LINCL. She exhibited a typical phenotype associated with LINCL, except she did not present spontaneous myoclonus, her symptoms occurrence was slower and developed focal sensory visual seizures. In addition, whole-exome sequencing identified a novel homozygous variant in CLN8, c.531G>T, resulting in p.Trp177Cys. Ultrastructural examination featured abundant lipofuscin deposits within mucosal cells, macrophages, and monocytes. We report a novel CLN8 mutation as a cause for NCL8 in a girl with developmental delay and epilepsy, cerebellar syndrome, visual loss, and progressive cognitive and motor regression. This case, together with an analysis of the available literature, emphasizes the existence of a continuous spectrum of CLN8-associated phenotypes rather than a sharp distinction between them.
Keyphrases
  • case report
  • endoplasmic reticulum
  • induced apoptosis
  • risk assessment
  • systematic review
  • breast cancer cells
  • oxidative stress
  • immune response
  • protein protein
  • endoplasmic reticulum stress
  • amino acid