Osteoporosis, bone & mineral

Efficacy and Toxicity of Calcitonin Treatment in Children with Cherubism: A Single-Center Cohort Study

J Bone Miner ResPublished Retrospective cohort study

Authors' conclusion

Calcitonin treatment seems effective and tolerable in treating actively progressing cherubism in children.

Study context

Population
Children with cherubism (three female, six male) during the progressive phase of the disease11 treatment courses
Participants
9 children
Outcome
Change in the volume of lesions during calcitonin treatment, assessed for the eight treatment courses with a minimal duration of 6 months
Follow-up
During treatment
Methodological detailssingle-centre, 11 treatment courses, mean duration 17.9 months (range <1 to 35, SD 10.8)
  • Single-centre
  • 11 treatment courses, mean duration 17.9 months (range <1 to 35, SD 10.8)

Main finding

As reported in the abstract

Change in cumulative lesion volume during treatment
  • Statistically significant reduction in mean cumulative lesion volume regardless of treatment duration
  • Average volume reduction highest in first half year of treatment, with gradual, ongoing reduction thereafter
  • 8 treatment courses with ≥6 months duration

Observational data: associations, not effects.

Secondary outcomes

Change in cumulative lesion volume after treatment cessation

Courses after which cumulative volume increased again but remained smaller than initial volume
6 of 7
  • 7 treatment courses with follow-up imaging

Safety

Treatment courses with adverse effects
73%
Grade 3 symptomatic hypocalcemia requiring hospitalization and early treatment termination
1
Most adverse effects mild and low grade
  • 11 treatment courses

Original publication

Efficacy and Toxicity of Calcitonin Treatment in Children with Cherubism: A Single-Center Cohort Study. J Bone Miner Res. Published 3 November 2023. doi:10.1002/jbmr.4922

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