Description of previous studies examining the use of antiresorptive agents in children with LCPD
Study | Study type | Diagnosis/ aetiology of avascular necrosis | Patients (n) | Age of patients | Severity at treatment initiation | Antiresorptive used | Other therapy used | Outcome measures | Follow-up length | Radiologic/ metabolic outcome | Functional outcome | Conclusion |
McQuade and Houghton15 | Case report | LCPD | 1 | 6.5 years | Unilateral; 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 months | Bone mineral density, mineral homeostasis, X-rays, pain and function. | 20 months following the start of treatment | Progressed 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 series | LCPD or slipped capital femoral epiphysis | 37 (17 with LCPD) | 10.8 years +/-2.76 years | LCPD diagnosed by standard radiologic criteria | Zoledronate, 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 months | PTH 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 report | LCPD | 1 | 9 years | >50% of epiphysis, severe (surgery recommended) | Denosumab: 60 mg, single injection | Varus osteotomy (1 year after onset and 7 months post-denosumab injection) | X-ray and MRI | 2 years 8 months | MRI 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 trial | LCPD | 100 | 5 years– 16 years | Lateral pillar >50% of the contralateral side | Zoledronate: five courses, 3-monthly doses of 0.025 mg/kg (12 month treatment) | Standard of care + non-weight bearing (wheel chair) first 6–12 months | Deformity index, bone density, MRI | Plan to follow-up to 24 months | Not yet completed. | Not yet completed. | Not completed, still underway |
Present study | Case report | LCPD | 1 | 6 years | Lateral pillar >50% involvement, bilateral. Catterall IV (right hip) | Pamidronate: nine courses, 1 mg/kg | Abductor tenotomies, broomstick casts (21 weeks total), Scottish Rite braces (1 year) | X-ray, bone density, MRI, pain | 11 years | Bone 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. |