[Analysis of genetic variant in a child with concomitant spinal muscular atrophy and Citrin protein deficiency]

Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2020 Aug 10;37(8):828-832. doi: 10.3760/cma.j.issn.1003-9406.2020.08.006.
[Article in Chinese]

Abstract

Objective: To explore the genetic basis for a child with concomitant spinal muscular atrophy (SMA) and Citrin protein deficiency.

Methods: The child was subjected to whole exome sequencing by using target sequence capture high-throughput sequencing. Candidate variants were verified by Sanger sequencing. The SMN genes of the patient were also analyzed through multiplex ligation-dependent probe amplification (MLPA).

Results: The patient was found to carry homozygous deletion of exons 7 and 8 of the SMN1 gene, for which his parents were both carriers. The patient also carried compound heterozygous variants c.1737G>A and IVS16ins3kbof the SLA25A13 gene, in addition with compound heterozygous variants c.948G>A and c.2693T>C of the POLG gene, for which his parents were carriers, too.

Conclusion: Variants of the SLC25A13 gene probably underlay the deficiency of Citrin protein, which may lead to neonatal intrahepatic cholestasis (NICCD). The patient also had SMA. The compound heterozygous variants c.948G>A and c.2693T>C of the POLG gene are likely to cause mitochondrial DNA deletion syndrome type 4A, though other types of mitochondrial disease cannot be excluded.

MeSH terms

  • Calcium-Binding Proteins / deficiency
  • Calcium-Binding Proteins / genetics*
  • Child
  • DNA Polymerase gamma / genetics
  • Homozygote
  • Humans
  • Mitochondrial Membrane Transport Proteins / genetics
  • Muscular Atrophy, Spinal / genetics*
  • Organic Anion Transporters / deficiency
  • Organic Anion Transporters / genetics*
  • Protein Deficiency / genetics*
  • Sequence Deletion
  • Survival of Motor Neuron 1 Protein / genetics

Substances

  • Calcium-Binding Proteins
  • Mitochondrial Membrane Transport Proteins
  • Organic Anion Transporters
  • SLC25A13 protein, human
  • SMN1 protein, human
  • Survival of Motor Neuron 1 Protein
  • citrin
  • DNA Polymerase gamma
  • POLG protein, human