Treatment of Chronic Nonbacterial Osteomyelitis with Bisphosphonates

Excerpt

A previously healthy 12-y-old girl with intermittent fever and progressive pain in her knee joints for two years was referred to rheumatology, poorly responding to the treatment of antibiotics, oral glucocorticoids (GC) combined with naproxen over 5 wk at another hospital. On physical examination, functional impairment, slight tenderness and swelling of the affected joints were found. Blood tests revealed mildly decreased count of WBC (3.0-3.64 × 10⁹/L) and elevation of ESR (30–70 mm/h). Whole-body magnetic resonance imaging (WB-MRI) suggested extensive bony edema at the metaphysis of lower extremity long bones (Fig. 1a); bone scintigraphy showed mild uptake in multiple joints symmetrically, and chronic inflammatory infiltrate with fibrosis was presented on biopsy. Considering these findings, a diagnosis of chronic nonbacterial osteomyelitis (CNO) was established; then methotrexate, naproxen, adalimumab with GC (0.5-1 mg/kg) were initiated. However, 3 mo later, the recurrent fever and intolerable pain drove her to authors’ institution again with worsened WB-MRI signals (Fig. 1b). Therefore, pamidronate (1 mg/kg, monthly) with sulfasalazine and thalidomide was employed. Consequently, clinical and radiological improvement was achieved significantly after 9 mo with 6 mo of pamidronate treatment (Fig. 1c). …