SAPHO syndrome and pamidronate revisited

M. Valls-Roc, M. Sanmarti, M. Salles, S. Holgado and A. Olive

Rheumatology Section, Hospital Universitari Germans Trias I Pujol, Carretera Canyet sn Badalona 08916, Spain

Correspondence: A. Olive, Rheumatology Section, Hospital Universitari Germans Trias Pujol, Ctrt Canyet sn Badalona 08916, Spain. E-mail: aolive{at}ns.hugtip.scs.es

SIR, We read with interest the paper of Amital et al. [1] and would like to comment on it. Since our unit was set up (1984) we have diagnosed 40 patients with the SAPHO syndrome. All the patients met the inclusion criteria of Benhamou et al. [2]. This was partially published elsewhere [3].

Pamidronate was indicated in those patients refractory to conventional therapy, namely non-steroidal anti-inflammatory drugs (NSAIDs), sulphasalazine and methotrexate. In all cases the indication for treatment was excruciating pain in the anterior chest wall (sternocostal, sternoclavicular or manubriosternal involvement). Six out of 40 were treated with intravenous pamidronate. The characteristics of the patients were: four female and two male; mean age 31 yr (range 14–49 yr); mean disease duration 5.5 yr. All of them were White. All patients were given 60 mg pamidronate intravenously on days 1, 7, 14, 28 and 56 [4]. All treatments were authorized by the Ministry of Health (compassionate use).

Clinical improvement was noted by all patients [visual analogue scale (VAS) for pain]; however, C-reactive protein (CRP) remained unchanged (Table 1). 99Tc bone scintigraphy was performed before and after the infusion. Interestingly, the bone uptake remained unchanged in all cases.


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TABLE 1. Characteristics of pamidronate treated patients

 
Adverse reactions were scarce, namely superficial phlebitis (venepuncture area) in two patients and fever during the infusion in one patient. Neither hypocalcaemia nor flu-like symptoms were present.

We would like to emphasize that the indication for pamidronate treatment was excruciating pain in the anterior chest wall, a hallmark of the SAPHO syndrome. The final dosage and the cadency of intravenous pamidronate was different from that used by Amital et al. [1]. Amital et al. administered 60 mg of intravenous pamidronate in cycles (one up to four). We followed the regime of Maksymowych et al. [4], i.e. intravenous pamidronate on days 1, 7, 14, 28 and 56.

Our impression is that intravenous pamidronate is a useful therapeutic agent for patients with refractory SAPHO syndrome. The amelioration is mainly clinical, as acute phase reactants and bone scintigraphy remained stable. Future studies should contemplate the dosage, cadency and duration of pamidronate treatment. Although SAPHO syndrome is an oddity, randomized studies are needed.

The authors have declared no conflicts of interest.

References

  1. Amital H, Applbaum YH, Aamar S, Daniel N, Rubinow A. SAPHO syndrome treated with pamidronate: an open label study of 10 patients. Rheumatology 2004;43:658–61.[Abstract/Free Full Text]
  2. Benhamou CL, Enjolras O, Delrieu F et al. Pseudoseptic arthritis and bacterid of Andrews. Rev Rhum Mal Ostearticul 1984;51:229–31.
  3. Olive A, Perez-Andres R, Rivas A et al. Síndrome SAPHO: estudio de 16 casos. Med Clin 1999;112:61–3.[ISI]
  4. Maksymowych WP, Lambert R, Jhangri GS et al. Clinical and radiological amelioration of refractory peripheral spondyloarthritis by pulse intravenous pamidronate therapy. J Rheumatol 2001;28:144–55.[ISI][Medline]
Accepted 5 October 2004





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