Unit of Rheumatology, Alcorcón Hospital Foundation, Madrid, Spain
Correspondence to: R. Almodóvar, Avda. Budapest s/n. 28922, Alcorcón Hospital Foundation, Madrid, Spain. E-mail: ralmodovar{at}fhalcorcon.es
SIR, Lymphoedema is an extra-articular manifestation rarely found in rheumatoid arthritis (RA) and juvenile idiopathic arthritis, and is quite rare in psoriatic arthritis (PsA) [14]. Literature research shows few published cases of lymphoedema: 30 cases in patients with RA and only eight cases in PsA [57]. The aetiology is unknown. Several hypotheses have been advanced on its pathogenesis in patients with RA, such as lymphangiitis, lymphatic obstruction, capillary permeability increase, abnormal fibrinolysis and other disorders related to lymphatic function and structure [4]. The diagnosis under suspicion is clinical, as one or more limbs have been observed to undergo painless swelling. Scintigraphy demonstrates existing lymphatic disorders [8]. Treatment for lymphoedema is inefficient and is usually limited to symptomatic treatment. In most cases of RA or PsA, introduction of disease-modifying drugs does not improve the oedema [3, 5, 911]. With regard to anti-TNF- therapy, infliximab, recently approved for treatment of ankylosing spondylitis (AS), has been found to be efficient on peripheral articular manifestations and on the axial skeleton. However, there is little information available on its efficacy for extra-articular manifestations. We describe the first case of a patient diagnosed with AS complicated with right upper limb lymphoedema who, after receiving treatment with infliximab, showed complete disappearance of the lymphoedema.
The patient was a 58-yr-old male with a background of gastric ulcer, venous thrombosis in the central nervous system and secondary osteoporosis. From the age of 15 he had presented a clinical picture compatible with AS, but this was not diagnosed until the age of 51. Initially, the patient was treated with non-steroid anti-inflammatory drugs (NSAIDs) and corticosteroids. In 1998, he developed a painless oedema in right upper limb. Lymphatic scintigraphy showed findings compatible with lymphoedema. In May 1999, treatment with sulphasalazine was begun and articular clinical results improved, but lymphoedema persisted and acute-phase reactants increased. In September 2000, methotrexate was added at a dose of 10 mg/week without improvement. In March 2001, previous therapies were discontinued and treatment with infliximab was begun at the dose of 5 mg/kg, repeating the dose at 2, 4 and 8 weeks. At 2 weeks, the patient experienced clinical improvement and acute-phase reactants had stabilized. At 12 weeks, the lymphoedema had disappeared completely. Treatment with infliximab was continued for 1 yr, after which it was discontinued when the patient was found to be asymptomatic. In September 2003, treatment was reinitiated due to symptomatic worsening of the axial skeleton.
Our patient is the first reported case presenting lymphoedema associated with AS in whom the lymphoedema disappeared completely after 3 months of treatment with infliximab. The way in which infliximab acts on lymphoedema is not known, but the drug is believed to act on the inflammatory response of the lymphatic vessels. Therefore, anti-TNF therapy can be considered for the treatment of extra-articular manifestations in AS, such as lymphoedema. Further experience is necessary to confirm the beneficial effect of infliximab and to learn how it acts on other extra-articular manifestations.
The authors have declared no conflicts of interest.
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