Table 1

Description of previous studies examining the use of antiresorptive agents in children with LCPD

StudyStudy typeDiagnosis/
aetiology of avascular necrosis
Patients (n)Age of patientsSeverity at treatment initiationAntiresorptive usedOther therapy usedOutcome measuresFollow-up lengthRadiologic/
metabolic outcome
Functional outcomeConclusion
McQuade and Houghton15 Case reportLCPD16.5 yearsUnilateral; lateral pillar group C.Zoledronte: 0.025 mg/kg administered 5 times in 9 months.Abductor tenotomy and broomstick casts (6 weeks), hip abduction brace 20 hours/day for 3 months) plus non-weight bearing for 6 monthsBone mineral density, mineral homeostasis, X-rays, pain and function.20 months following the start of treatmentProgressed to remodelling phase on X-ray 11 month post completion of therapy, and from laterall pillar group C to group B. No abnormalities on renal ultrasound. Mild decreases in serum calcium noted postinfusion.Pain rated zero 2 weeks postinfusion. At the time of reporting, contact sports were still restricted with follow-up planned in 6 months.Zolendronate therapy for LCPD produced a positive radiologic response and positive responses for function and pain control. There was also a positive emotional response from family regarding the treatment.
Johannesen et al 16 Prospective case seriesLCPD or slipped capital femoral epiphysis37 (17 with LCPD)10.8 years +/-2.76 yearsLCPD diagnosed by standard radiologic criteriaZoledronate, first dose 0.0125 mg/kg; subsequent doses 0.025 mg/kg (second dose given 6 weeks later, subsequently at 12 week intervals): for at least 12 months Average number of treatments per patient was 6.Non-weight bearing for the first 6 months of Zoledronate therapy; 400 IU vitamin D and calcium supplementation if intake
<recommended.
Bone mineral density, bone morphometry/markers of turnover, and mineral homeostasis.18 monthsPTH increased during treatment, 25OHD unchanged. Greatest increase in Bone mineral density in the lumbar spine of children with LCPD.
No radiologic (X-ray/MRI) outcome of hip reported.
Not reported.Zoledronate increased bone mineral density in children with avascular necrosis without any symptomatic side effects. More trials are warranted.
Meiss et al 26 Case reportLCPD19 years>50% of epiphysis, severe (surgery recommended)Denosumab: 60 mg, single injectionVarus osteotomy (1 year after onset and 7 months post-denosumab injection)X-ray and MRI2 years 8 monthsMRI 2 years post diagnosis showed well contained head with re-ossification.The boy could comfortably carry out daily activities with no pain or limp. Normal hip motion.Denosumab may have had positive effect on the outcome, but the patient was also treated with an osteotomy.
Jamil et al 17 (currently underway)Randomised clinical trialLCPD1005 years– 16 yearsLateral pillar >50% of the contralateral sideZoledronate: five courses, 3-monthly doses of 0.025 mg/kg (12 month treatment)Standard of care + non-weight bearing (wheel chair) first 6–12 monthsDeformity index, bone density, MRIPlan to follow-up to 24 monthsNot yet completed.Not yet completed.Not completed, still underway
Present studyCase reportLCPD16 yearsLateral pillar >50% involvement, bilateral. Catterall IV (right hip)Pamidronate: nine courses, 1 mg/kgAbductor tenotomies, broomstick casts (21 weeks total), Scottish Rite braces (1 year)X-ray, bone density, MRI, pain11 yearsBone mineral density (DEXA) Z-scores increased alongside treatment. Femoral heads show complete improvement with normalised joint spaces (X-ray/MRI).No functional limitations, no pain, normal hip motion.Pamidronate therapy showed positive effect on LCPD and likely helped avoid surgery. No negative effects seen in a case study at 11 years.