Intraventricular Cerliponase Alfa Treatment in a Patient with Advanced Neuronal Ceroid Lipofuscinosis Type 2

Intern Med. 2024 Jun 15;63(12):1807-1812. doi: 10.2169/internalmedicine.2563-23. Epub 2023 Nov 6.

Abstract

Neuronal ceroid lipofuscinosis type 2 (CLN2) is an autosomal recessive lysosomal disease caused by decreased activity of the enzyme tripeptidyl peptidase 1 (TPP1) due to pathogenic variants in the TPP1 gene. Cerliponase alfa, a recombinant proenzyme form of TPP1, has shown efficacy in preventing motor and language function decline in early-stage CLN2. However, the safety and effects of this therapy in advanced-stage CLN2 are unclear. We herein report a case of intraventricular cerliponase alfa treatment for over a year in a patient with advanced-stage CLN2. The results suggest the safety and potential efficacy of treatment at an advanced stage of CLN2.

Keywords: cerliponase alfa; motor language score; neuronal ceroid lipofuscinosis; seizure; tripeptidyl peptidase 1.

Publication types

  • Case Reports

MeSH terms

  • Aminopeptidases* / genetics
  • Dipeptidyl-Peptidases and Tripeptidyl-Peptidases*
  • Humans
  • Male
  • Neuronal Ceroid-Lipofuscinoses* / complications
  • Neuronal Ceroid-Lipofuscinoses* / drug therapy
  • Neuronal Ceroid-Lipofuscinoses* / genetics
  • Recombinant Proteins / administration & dosage
  • Recombinant Proteins / therapeutic use
  • Serine Proteases* / genetics
  • Treatment Outcome
  • Tripeptidyl-Peptidase 1*
  • Young Adult

Substances

  • Aminopeptidases
  • cerliponase alfa
  • Dipeptidyl-Peptidases and Tripeptidyl-Peptidases
  • Recombinant Proteins
  • Serine Proteases
  • TPP1 protein, human
  • Tripeptidyl-Peptidase 1

Supplementary concepts

  • Ceroid Lipofuscinosis, Neuronal, 2