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Serum brain-type creatine kinase increases in children with osteogenesis imperfecta during neridronate treatment

Patrizia D'Eufemia1, Roberto Finocchiaro1, Ciro Villani2

  • 1Department of Pediatrics, "Sapienza", University of Rome, Rome, Italy.

Pediatric Research
|February 13, 2014
PubMed

Insights

Serum creatine kinase-brain isoform (Ckbb) levels increase during bisphosphonate treatment in children with osteogenesis imperfecta (OI). This suggests suppressed osteoclast activity, potentially aiding in preventing overtreatment in pediatric patients.

Area of Science:

  • Biochemistry
  • Pediatric Endocrinology
  • Bone Biology

Background:

  • Creatine kinase (Ck) facilitates energy transfer, with the brain isoform (Ckbb) crucial for osteoclast function in bone resorption.
  • Elevated serum Ckbb is observed in osteopetrosis and bisphosphonate (BP)-induced conditions.
  • Bisphosphonates (BPs) are standard treatment for osteogenesis imperfecta (OI), inhibiting bone resorption by osteoclasts.

Purpose of the Study:

  • To investigate changes in serum total Ck and Ckbb activity in prepubertal children with type I OI undergoing bisphosphonate therapy.
  • To assess the correlation between serum Ckbb levels and bone resorption markers during treatment.

Main Methods:

  • Serum Ck and isoform activity were measured in 18 children with type I OI before and during neridronate infusions.
  • Statistical analysis was performed to determine significant changes and correlations.

Main Results:

  • Basal serum Ckbb levels were slightly elevated compared to controls.
  • Serum Ckbb levels progressively increased after neridronate treatment, with significant increments observed after infusions.
  • An inverse correlation was noted between basal serum Ckbb and serum CTx levels.

Conclusions:

  • Increased serum Ckbb supports the hypothesis that it reflects osteoclast suppression or failure.
  • The findings suggest that monitoring serum Ckbb could help prevent overtreatment with long-acting bisphosphonates in pediatric OI patients.
Abstract

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