[Analysis of phenotype and genotype in a family with late infantile metachromatic leukodystrophy]

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2014 Oct;31(5):615-8. doi: 10.3760/cma.j.issn.1003-9406.2014.01.017.
[Article in Chinese]

Abstract

Objective: To study genotype-phenotype correlation of a family with late infantile metachromatic leukodystrophy(MLD).

Methods: Clinical data were collected and ARSA gene was tested by PCR and sequencing in a pedigree.

Results: The male proband onset with walking dysfunction at 19 months, arylsulfatase A activity of leucocyte from his peripheral blood was 20.2 nmol/mg.17h, and his cranial MRI showed wildly symmetrical demyelination. Homozygosis for novel c.622delC (p.His208Metfs46X) in exon 3 of ARSA gene was identified in proband, and heterozygous for the same mutation in parents and grandma of the proband.

Conclusion: Late infantile metachromatic leukodystrophy is characterized by rapid and progressive regression of neuropsychiatric and motor development. There is a significant correlation between the mutation of c.622delC(p.His208Metfs*46) in the ARSA gene and the phenotype presenting as O/O patients.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Base Sequence
  • Cerebroside-Sulfatase / deficiency
  • Cerebroside-Sulfatase / genetics*
  • DNA Mutational Analysis
  • Family Health
  • Female
  • Genetic Predisposition to Disease / genetics*
  • Genotype
  • Humans
  • Infant
  • Leukodystrophy, Metachromatic / diagnostic imaging
  • Leukodystrophy, Metachromatic / enzymology
  • Leukodystrophy, Metachromatic / genetics*
  • Magnetic Resonance Imaging
  • Male
  • Mutation*
  • Pedigree
  • Phenotype
  • Polymerase Chain Reaction
  • Radiography
  • Sequence Deletion

Substances

  • Cerebroside-Sulfatase