Rheumatology Advance Access originally published online on August 15, 2003
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Rheumatology 2004; 43: 229-233
© British Society for Rheumatology 2003; all rights reserved
Clinical |
Safety and efficacy of iloprost for the treatment of ischaemic digits in paediatric connective tissue diseases
Pediatric Rheumatology Unit, University of Padua, 1Istituti Clinici di Perfezionamento, University of Milan, 2Hospital ‘G. Pini’, University of Milan, 3Children's Hospital ‘Meyer’ Florence and 3Children's Hospital ‘G. Gaslini’, Genoa, Italy
Correspondence to: F. Zulian, Paediatric Rheumatology Unit, Department of Paediatrics, Via Giustiniani 3, 35128 Padova, Italy. E-mail: zulian{at}pediatria.unipd.it
Objective. We analysed our experience with the use of iloprost for the treatment of critical ischaemic digits (ID) in children with connective tissue diseases (CTD) in order to assess its safety and efficacy.
Methods. This was a retrospective analysis of paediatric patients with CTD who were treated with iloprost for critical ID resistant to conventional therapy. Information on demographics, clinical and laboratory features, the regimen of iloprost treatment and outcome were collected.
Results. Fifteen patients (10 female, five male) treated one or more times with iloprost were included (total of 19 treatments). Six had juvenile systemic sclerosis, five had systemic lupus erythematosus, three had mixed connective tissue disease and one had cutaneous polyarteritis nodosa. Thirteen patients were already taking calcium channel blockers with no improvement; in two patients ID were the presenting signs of the disease without prior treatment. Eleven patients had more than two fingers involved; one child had involvement of all 10 fingers. Normal digital blood flow was achieved in 74% of treatments and significant improvement was noted in 26%. Fingertip necrosis was present in 11 patients (14 treatments). It healed completely in seven, improved in one and remained unchanged in six. Raynaud's phenomenon (RP) was present in 14 patients (17 treatments): in two no RP attack occurred during the follow-up period, eight improved both in the number of attacks per week and in the duration of each attack. Complete pain relief was observed in 10/17 treatments (59%) and there was a significant decrease in pain in the remaining seven. No major side-effects or withdrawal symptoms were reported. Minor side-effects reported include reversible headache (seven patients), hypotension or irritability (three), nausea/vomiting (two) and injection site reaction (one).
Conclusions. Iloprost appears to be a safe and effective treatment for ischaemic digits and digital ulcers in children with CTD. In conjunction with immunosuppressive drugs, it has a potential role in preventing irreversible complications, such as digital gangrene and amputation.
KEY WORDS: Iloprost, Necrotizing vasculopathy, Ischaemic digits, Paediatric CTD.
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