Rheumatology Advance Access originally published online on July 6, 2004
Rheumatology 2004 43(10):1246-1251; doi:10.1093/rheumatology/keh295
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Rheumatology Vol. 43 No. 10 © British Society for Rheumatology 2004; all rights reserved
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Pamidronate in the treatment of childhood SAPHO syndrome
Royal Liverpool Children's NHS Trust, Eaton Road, Liverpool L12 2AP, UK.
Correspondence to: M. W. Beresford, Department of Rheumatology, Royal Liverpool Children's NHS Trust, Eaton Road, Liverpool L12 2AP, UK. E-mail: m_beresford{at}yahoo.com
Background. SAPHO syndrome is increasingly recognized within the paediatric population. Conventional therapeutic approaches have often not been effective. Pamidronate is a second-generation bisphosphonate that affects bone turnover and demonstrates anti-inflammatory properties. In small case series it has given symptomatic relief to adults with this condition.
Objectives. To report the clinical experience with pamidronate in childhood SAPHO syndrome.
Methods. A retrospective observational study of all children with SAPHO syndrome treated with pamidronate between 1996 and 2003 at a tertiary rheumatology centre. The standard dosing regime for pamidronate was 1 mg/kg to a maximum of 30 mg, administered daily for three consecutive days, repeated 3-monthly as required. Response to treatment was determined by clinical observation, patient subjective response and reduction in other treatments
Results. Seven girls were treated, with a median (range) age at diagnosis of 11 yr (9–15 yr). All patients demonstrated a beneficial clinical response, with relief of pain, increased activity and improved well-being. Subsequent courses of pamidronate were used in all patients. Other medications including corticosteroids and methotrexate could subsequently be stopped. Transient symptoms were associated with the initial course of pamidronate in some patients. No serious adverse events were reported.
Conclusions. Pamidronate was associated with a marked improvement in function and well-being, and a reduction of pain and use of other medications in all patients, with no significant adverse effects. This study represents preliminary clinical data. A prospective multicentre study is necessary to assess the role and long-term safety of pamidronate in the management of childhood SAPHO syndrome
KEY WORDS: SAPHO, CRMO, Pamidronate, Childhood
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