Table 2. Data on children and adolescents with osteogenesis imperfecta treated with denosumab reported in the literature.

Study	Year	Patient age	Dose and interval of denosumab	Duration of treatment	Effect of treatment	Adverse events during treatment	Adverse events after treatment
Semler et al. (6)	2012	4 boys age 5–18 years (type 6)	1 mg/kg every 8–12 weeks	24 months	Increased BMD, improved pain	Mild hypocalcemia (1 patient)	Unknown
Hoyer-Kuhn et al. (12)	2014	4 boys age 5–18 years (type 6)	1 mg/kg every 8–12 weeks	24 months	Increased BMD, improved pain	Mild hypocalcemia (1 patient)	Unknown
Ward et al. (13)	2016	1 child age 2 years (type 6)	1 mg/kg every 4–12 weeks	12 months	Increased BMD, improved mobility, more osteoclasts formed	Severe hypercalcemia	None
Hoyer-Kuhn et al. (14)	2016	10 children age 5–11 years (types 1, 3, and 4)	1 mg/kg every 12 weeks	12 months	Increased BMD and increased height; bone pain did not change	Mild hypocalcemia (1 patient)	Mild hypercalcemia
Uehara et al. (15)	2017	1 child age 14 years	Every 6 months	24 months	Increased BMD	None	Unknown
Trejo et al. (16)	2018	4 children age 1.9–9 years (type 6)	1 mg per kg every 3 months	Mean 24 months	Increased BMD	Mild hypercalcemia and persistent hypercalciuria, nephrocalcinosis, rapid bone loss	None
Hoyer-Kuhn et al. (17)	2019	10 children, mean age 8.6 (6.16–12.13 years) (types 1, 3, and 4)	1 mg per kg every 20.3 weeks (depending on the individual urinary excretion course of deoxypyridinoline)	53.04 weeks (± SD 6.30)	Increased BMD	Arthralgia, muscle pain, symptomatic hypercalciuria	Symptomatic hypercalciuria

SD, standard deviation; BMD, bone mineral density.