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Rheumatology Advance Access published online on July 6, 2004
Rheumatology, doi:10.1093/rheumatology/keh295
Rheumatology © British Society for Rheumatology 2004; all rights reserved
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Received March 22, 2004
Accepted June 4, 2004

Original Papers

Pamidronate in the treatment of childhood SAPHO syndrome

C. Kerrison 1, J. E. Davidson 1, A. G. Cleary 1, M. W. Beresford 1*

1 Royal Liverpool Children's NHS Trust, Eaton Road, Liverpool L12 2AP, UK

* To whom correspondence should be addressed. E-mail: m_beresford{at}yahoo.com.


  Abstract

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

Keywords: SAPHO; CRMO; Pamidronate; Childhood.
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