Nephrotic syndrome and thrombotic microangiopathy caused by cobalamin C deficiency

Pediatr Nephrol. 2015 Jul;30(7):1203-6. doi: 10.1007/s00467-015-3110-x. Epub 2015 Apr 18.

Abstract

Background: Cobalamin C (CblC) defects are inherited autosomal recessive disorders of vitamin B12 metabolism due to mutations in the MMACHC gene. Renal manifestations include thrombotic microangiopathy (TMA), acute or chronic renal failure, tubulointerstitial nephritis, and proximal renal tubular acidosis. However, reports about glomerular pathologies are scarce.

Case report: A 4-year-old boy presented with nephrotic syndrome, arterial hypertension, and chronic anemia but no signs of hemolysis. Renal biopsy showed TMA with ischemic glomerular collapse, foot process effacement, and tubulointerstitial fibrosis. Elevated serum levels of homocysteine suggested a cobalamin C disorder. This was confirmed by the identification of compound heterozygous mutations in the MMACHC gene. Initial therapy consisted of antihypertensive treatment including angiotensin converting enzyme inhibitor (ACEi) leading to blood pressure control and a significant reduction of proteinuria. After a definite diagnosis of CblC deficiency, hydroxocobalamin was introduced. Thereafter, homocysteine levels decreased, anemia resolved, and a further decline of proteinuria with normalization of serum protein levels was noted. Renal function remained stable.

Conclusions: Although uncommon, the clinical picture of CblC defects may be ruled by nephrotic syndrome mimicking glomerulonephritis, minimal change disease, or primary focal and segmental glomerulosclerosis. Key to a correct diagnosis is elevated serum levels of homocysteine, and a definite diagnosis can be confirmed by genetic testing.

Publication types

  • Case Reports

MeSH terms

  • Anemia / etiology
  • Angiotensin-Converting Enzyme Inhibitors / therapeutic use
  • Biopsy
  • Carrier Proteins / genetics
  • Child, Preschool
  • Homocysteine / blood
  • Humans
  • Hydroxocobalamin / therapeutic use
  • Hypertension, Renal / drug therapy
  • Hypertension, Renal / etiology
  • Hypertension, Renal / pathology
  • Kidney / pathology
  • Male
  • Nephrotic Syndrome / drug therapy
  • Nephrotic Syndrome / etiology*
  • Nephrotic Syndrome / pathology
  • Oxidoreductases
  • Thrombotic Microangiopathies / drug therapy
  • Thrombotic Microangiopathies / etiology*
  • Thrombotic Microangiopathies / pathology
  • Vitamin B 12 / metabolism*
  • Vitamin B 12 Deficiency / complications*
  • Vitamin B 12 Deficiency / genetics
  • Vitamin B 12 Deficiency / pathology

Substances

  • Angiotensin-Converting Enzyme Inhibitors
  • Carrier Proteins
  • Homocysteine
  • MMACHC protein, human
  • Oxidoreductases
  • Vitamin B 12
  • Hydroxocobalamin