Clinical and biochemical outcomes in cobalamin C deficiency with use of high-dose hydroxocobalamin in the early neonatal period

Am J Med Genet A. 2022 Jun;188(6):1831-1835. doi: 10.1002/ajmg.a.62687. Epub 2022 Feb 14.

Abstract

This case report describes a patient with early-onset cobalamin C deficiency who was started on treatment with high-dose parenteral hydroxocobalamin after diagnosis at 13 days of life. Prior to diagnosis, initial presenting symptoms included poor feeding, lethargy, apneic episodes, hypothermia, and hypotonia; these symptoms resolved after initiation of medication. Methylmalonic acid and homocysteine levels were trended and significantly improved with treatment. She was maintained on 2 mg/kg/day dosing of hydroxocobalamin. No adverse effects to treatment were observed. At the time of this report, the patient was 19 months of age; she had not manifested common findings of early-onset cobalamin C deficiency, including microcephaly, poor feeding, growth abnormalities, hypotonia, seizures, maculopathy, or neurodevelopmental delay. This report suggests that early initiation of high-dose hydroxocobalamin is safe and effective.

Keywords: MMACHC; biochemical genetics; cobalamin C deficiency; hydroxocobalamin; inborn errors of metabolism.

Publication types

  • Case Reports

MeSH terms

  • Amino Acid Metabolism, Inborn Errors* / diagnosis
  • Amino Acid Metabolism, Inborn Errors* / drug therapy
  • Female
  • Humans
  • Hydroxocobalamin / therapeutic use
  • Infant, Newborn
  • Methylmalonic Acid
  • Muscle Hypotonia / drug therapy
  • Vitamin B 12 Deficiency* / diagnosis
  • Vitamin B 12 Deficiency* / drug therapy

Substances

  • Methylmalonic Acid
  • Hydroxocobalamin