[Niemann-Pick disease type C caused by NPC1 mutation in a case]

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2019 May 10;36(5):480-483. doi: 10.3760/cma.j.issn.1003-9406.2019.05.016.
[Article in Chinese]

Abstract

Objective: To delineate the clinical and genetic features of a Chinese boy suspected for Niemann-Pick disease type C.

Methods: The patient underwent clinical examination and was subjected to next generation sequencing. Suspected mutations were validated by Sanger sequencing. Potential impact of the novel mutation was predicted by SIFT, PolyPhen-2 and MutationTaster software.

Results: The child has featured hepatosplenomegaly, increased direct bilirubin, jaundiced skin and liver damage. DNA sequencing showed that he has carried compound heterozygous mutations of NPC1 gene, namely c.2728G<A (p.G910S) and c.269C>G (p.P90R), which were inherited from his mother and father, respectively. The c.2728G>A (p.G910S) mutation was previously reported, while the c.269C>G (p.P90R) was a novel mutation.

Conclusion: The child has suffered from Niemann-Pick disease type C due to mutations of NPC1 gene. Above finding has enriched the spectrum of NPC1 mutations and provided a basis for genetic counseling and prenatal diagnosis.

Publication types

  • Case Reports

MeSH terms

  • Asian People
  • Bilirubin
  • Carrier Proteins / genetics*
  • Child
  • High-Throughput Nucleotide Sequencing
  • Humans
  • Intracellular Signaling Peptides and Proteins
  • Male
  • Membrane Glycoproteins / genetics*
  • Mutation
  • Niemann-Pick C1 Protein
  • Niemann-Pick Disease, Type C* / genetics

Substances

  • Carrier Proteins
  • Intracellular Signaling Peptides and Proteins
  • Membrane Glycoproteins
  • NPC1 protein, human
  • Niemann-Pick C1 Protein
  • Bilirubin