CoQ10-related sustained remission of proteinuria in a child with COQ6 glomerulopathy-a case report

Pediatr Nephrol. 2018 Dec;33(12):2383-2387. doi: 10.1007/s00467-018-4083-3. Epub 2018 Sep 19.

Abstract

Background: Treatment of steroid resistant nephrotic syndrome is still a challenge for physicians. There are a growing number of studies exploring genetic background of steroid-resistant glomerulopathies.

Case diagnosis/treatment: We present the case of a 4-year-old girl with steroid-resistant glomerulopathy due to a COQ6 defect with no additional systemic symptoms. The disease did not respond for second-line therapy with calcineurin inhibitor, but it remitted completely after oral treatment with 30 mg/kg/d of coenzyme Q10 (CoQ10). The patient was identified to be a compound heterozygote for two pathogenic variants in COQ6 gene: a known missense substitution c.1078C > T (p.R360W) and a novel frameshift c.804delC mutation. After 12 months of CoQ10 therapy, the child remains in full remission, her physical development accelerated, frequent respiratory airways diseases subsided.

Conclusions: Genetic assessment of children with steroid-resistant nephrotic proteinuria enables therapy optimization. Proteinuria caused by a COQ6 gene defect can be successfully treated with CoQ10.

Keywords: Child; Genetics; Nephropathy; Primary coenzyme Q10 deficiency; Treatment; Ubiquinone.

Publication types

  • Case Reports

MeSH terms

  • Angiotensin-Converting Enzyme Inhibitors / administration & dosage
  • Calcineurin Inhibitors / administration & dosage
  • Child, Preschool
  • Drug Therapy, Combination / methods
  • Female
  • Genetic Testing
  • Glomerulosclerosis, Focal Segmental / diagnosis
  • Glomerulosclerosis, Focal Segmental / drug therapy*
  • Glomerulosclerosis, Focal Segmental / genetics
  • Glomerulosclerosis, Focal Segmental / urine
  • Glucocorticoids / administration & dosage
  • Heterozygote
  • Humans
  • Mutation
  • Nephrosis, Lipoid / diagnosis
  • Nephrosis, Lipoid / drug therapy*
  • Nephrosis, Lipoid / genetics
  • Nephrosis, Lipoid / urine
  • Proteinuria / diagnosis
  • Proteinuria / drug therapy*
  • Proteinuria / genetics
  • Proteinuria / urine
  • Treatment Outcome
  • Ubiquinone / administration & dosage
  • Ubiquinone / analogs & derivatives*
  • Ubiquinone / genetics

Substances

  • Angiotensin-Converting Enzyme Inhibitors
  • Calcineurin Inhibitors
  • Glucocorticoids
  • ubiquinone 6
  • Ubiquinone
  • coenzyme Q10