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Rheumatology 2003; 42: 1273-1274
© 2003 British Society for Rheumatology
Letter to the Editor |
Pancytopenia in a patient with scleroderma treated with infliximab
Rheumatology, LSUHSC, New Orleans, LA, USA
Correspondence to:
L. R. Espinoza. E-mail: yaminimenon{at}msn.com
SIR, We read with interest the report by Marchesoni et al. [1] of life-threatening reversible bone marrow toxicity in a rheumatoid arthritis patient treated initially with methotrexate and later switched to leflunomide plus infliximab. The authors suggest that leflunomide may have been responsible for the pancytopenia in their case.
We took care of a 45-yr-old female nurse with scleroderma diagnosed 5 yr earlier when she presented with arthralgias, sclerodactyly and a history consistent with Raynaud’s phenomenon. She was anaemic (haemoglobin 10.5 gm/dl), had positive antinuclear antibodies (ANA) at a titre of 1:160, and had renal insufficiency (blood urea nitrogen 37 mg/dl, creatinine 1.7 mg/dl). Initial therapy included D-penicillamine 125 mg daily and enalapril 10 mg twice daily. Three months later she developed renal crisis and had worsening of renal function that required haemodialysis for approximately 14 months. Renal function stabilized with a serum creatinine level at around 3 mg/dl. She did well for the next 2 yr, until she suddenly developed severe dyspnoea that worsened over the ensuing few weeks. Chest radiographs showed interstitial lung disease and pulmonary function tests showed a restrictive pattern. Transthoracic echocardiography showed a pulmonary artery pressure of 45 mmHg. Her underlying renal failure limited the use of cytotoxic and immunosuppressive agents. Therefore, infliximab therapy was discussed with the patient and, after agreeing to it, she received an infusion of infliximab (300 mg intravenously over 2 h). Six days after receiving the infusion, the patient reported a late hypersensitivity reaction that lasted 3 days and was characterized by sore throat, malaise and severe arthralgias, and she refused further infliximab infusions. Two weeks later she was hospitalized with nausea, vomiting and progressive weakness. She was found to be hypotensive and dehydrated. Blood tests revealed pancytopenia [white blood cell count (WBC) 2.3 x 103/mm3, haemoglobin (Hb) 6.8 g/dl, haematocrit (Hct) 17.5, platelets 37 000/mm3]. Prior to infliximab therapy her WBC was 8.0 x 103/mm3, Hb 11 g/dl, Hct 35 and platelet count 200 000/mm3. She developed fever up to 102°F (38.9°C) and abdominal pain over the next 24 h, and ascitis was present. Peritoneal fluid was cloudy, with 322 red blood cells/mm3, 4950 white blood cells/mm3 and 96% neutrophils. Fluid cultures later grew Candida albicans. According to the patient’s wishes, only blood transfusions, antibiotics and supportive care were given, and she expired the next day. No autopsy was done.
Tumour necrosis factor (TNF) inhibitors are being investigated in the treatment of a variety of rheumatic disorders, including scleroderma [2, 3]. Early results with etanercept indicate marginal clinical improvement, especially of skin involvement. Due to the severity of the patient’s clinical picture, infliximab therapy was given. This was followed by pancytopenia and fungal infection. She was not receiving any other therapy that may have induced her haematological complication, which led us to implicate infliximab as an important contributor. This case should be added to the cases of existent pancytopenia already reported in association with anti-TNF-
therapy [4, 5].
The authors have declared no conflicts of interest.
References
- Marchesoni A, Arreghini M, Panni B, Battafarano N, Uziel L. Life threatening reversible bone marrow toxicity in a rheumatoid arthritis patient switched from leflunomide to infliximab. Rheumatology 2003;42:193–4
[Free Full Text] - Ellman MH, McDonald PA, Hayes FA. Etanercept as treatment for diffuse scleroderma: a pilot study. Arthritis Rheum 2000;43(Suppl.):S392
- Fitzcharles MA, Clayton D, Menard HA. The use of infliximab in academic rheumatology practice: an audit of early clinical experience. J Rheumatol 2002;29:2525–30
[Abstract/Free Full Text] - Day R. Adverse reactions of TNF inhibitors in rheumatoid arthritis. Lancet 2002;359:540–1[CrossRef][Web of Science][Medline]
- Steensma DP, Mesa RA, Li CY, Gray L, Tefferi A. Etanercept, a soluble tumor necrosis factor receptor, palliates constitutional symptoms in patients with myelofibrosis with myeloid metaplasia: results of a pilot study. Blood 2002;99:2252–4
[Abstract/Free Full Text]
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