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.
Abstract:
Pelizaeus-Merzbacher-like disease (PMLD) is an autosomal recessive hypomyelinating leukodystrophy with clinical symptoms and imaging manifestations similar to those of Pelizaeus-Merzbacher disease (PMD), an X-linked recessive hypomyelinating leukodystrophy. Typical manifestations of PMLD are nystagmus, dysmyotonia, ataxia, progressive motor dysfunction, and diffuse leukodystrophy on magnetic resonance imaging (MRI). This report identified novel mutations in NCP1 causing PMLD. A 7-month-old male patient was referred to our hospital because he could not lift his head until that time. He had symptoms including congenital nystagmus, hypotonia, and developmental delay. According to the MRI scan, there were signs of leukodystrophy. According to the clinical manifestations and the results of whole-exome sequencing (compound heterozygote mutations in NPC1 (p. G911S, c2731G>A and p. D128H, c382G>C)), the diagnosis of PMLD was considered, and his parents were determined to be carriers of mutant genes. He began rehabilitation training at the age of 1 year old. After 5 years of training, he was still experiencing global developmental delay, equivalent to the developmental level of a nine-month-old child. PMLD is a disease that seriously affects the quality of life of children and can result from mutations in different genes. In this report, we expand the gene spectrum of PMLD and suggest early genetic counselling for suspected patients and their patients.
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.
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