Specific biochemical markers of bone metabolism and cytokine study confirm the diagnosis of malignant infantile osteopetrosis at birth using cord blood sample

Pathology. 2005 Feb;37(1):51-5. doi: 10.1080/00313020400024725.

Abstract

Aims: To investigate the serum creatine kinase isoenzyme pattern, specific biochemical markers of bone metabolism, and cytokines in a Chinese family with osteopetrosis, and correlate abnormalities with the pathophysiology of this condition.

Methods: A Chinese female baby was diagnosed with malignant infantile osteopetrosis at the age of 3 weeks by clinical history and biochemical investigations. We studied the laboratory and radiological manifestations of this index case and her family members.

Results: Serum CK-BB fraction of our index patient was elevated to 18.0% (normal 1.6-7.6%). Her biochemical markers of bone resorption including serum C-terminal telopeptide concentration and urine N-terminal telopeptide to creatinine ratio were decreased to 0.54 microg/L (normal 0.72-1.56 microg/L) and 159 x 10(-6) (normal 372-900 x 10(-6)), respectively. Serum cytokines including soluble receptor activator of nuclear factor kappa-B ligand (sRANKL) concentration was suppressed to 0.11 pmol/L (normal 0.23-0.82 pmol/L) and osteoprotegerin (OPG) concentration was 4.9 pmol/L (normal 2.8-4.9 pmol/L), resulting in an elevated OPG to sRANKL ratio of 44.5 (normal 3.8-19.4) in favour of bone formation.

Conclusions: If left untreated, this condition is usually fatal within the first year of life. With early diagnosis, management including bone marrow transplantation can be planned ahead and will result in a better survival.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Biomarkers
  • Bone Marrow Transplantation
  • Bone and Bones / metabolism*
  • Carrier Proteins / blood
  • China
  • Creatine Kinase / blood*
  • Cytokines
  • Female
  • Fetal Blood*
  • Glycoproteins / blood
  • Humans
  • Infant, Newborn
  • Isoenzymes / blood*
  • Male
  • Membrane Glycoproteins / blood
  • Osteopetrosis / blood*
  • Osteopetrosis / diagnosis*
  • Osteopetrosis / therapy
  • Osteoprotegerin
  • RANK Ligand
  • Receptor Activator of Nuclear Factor-kappa B
  • Receptors, Cytoplasmic and Nuclear / blood
  • Receptors, Tumor Necrosis Factor

Substances

  • Biomarkers
  • Carrier Proteins
  • Cytokines
  • Glycoproteins
  • Isoenzymes
  • Membrane Glycoproteins
  • Osteoprotegerin
  • RANK Ligand
  • Receptor Activator of Nuclear Factor-kappa B
  • Receptors, Cytoplasmic and Nuclear
  • Receptors, Tumor Necrosis Factor
  • TNFRSF11A protein, human
  • TNFRSF11B protein, human
  • TNFSF11 protein, human
  • Creatine Kinase