Novel Mutations in NPC1 are Associated with Pelizaeus-Merzbacher-Like Disease: A Case Report

Hongling Fu1,2, Qiu Wang2,3, Hanmin Liu1,2

  • 1Department of Pediatrics, West China Second University Hospital, Sichuan University, Chengdu, Sichuan, People's Republic of China.

Insights

Pelizaeus-Merzbacher-like disease (PMLD) is a genetic disorder affecting myelin. This study identifies novel NPC1 gene mutations causing PMLD, expanding the known genetic causes of this severe condition.

Area of Science:

  • Neurology
  • Genetics
  • Pediatrics

Background:

  • Pelizaeus-Merzbacher-like disease (PMLD) is a rare, autosomal recessive hypomyelinating leukodystrophy.
  • It shares clinical and imaging features with X-linked Pelizaeus-Merzbacher disease (PMD).
  • Key symptoms include nystagmus, ataxia, motor dysfunction, and MRI-detected leukodystrophy.

Observation:

  • A 7-month-old male infant presented with congenital nystagmus, hypotonia, and developmental delay.
  • MRI revealed diffuse leukodystrophy.
  • Whole-exome sequencing identified compound heterozygous mutations in the NPC1 gene (p.G911S and p.D128H).

Findings:

  • The identified compound heterozygous mutations in NPC1 (c.2731G>A and c.382G>C) are novel causes of PMLD.
  • The patient exhibited persistent global developmental delay despite early rehabilitation.
  • Parents were confirmed as carriers of the identified NPC1 mutations.

Implications:

  • This study expands the genetic spectrum of genes associated with PMLD.
  • Early genetic counseling is crucial for families with suspected PMLD.
  • Understanding novel genetic mutations aids in diagnosis and potential therapeutic strategies for hypomyelinating leukodystrophies.