Chao Sheau-Chiou, Yang Chao-Chun, Lee Julia Yu-Yun
Department of Dermatology, College of Medicine, National Cheng Kung University, Tainan, Taiwan.
Ann Pharmacother. 2005 Sep;39(9):1552-6. doi: 10.1345/aph.1G105. Epub 2005 Aug 2.
To report a rare case of combined hypersensitivity syndrome and pure red cell aplasia (PRCA) following allopurinol therapy.
A 43-year-old woman with underlying mesangioproliferative glomerulonephritis developed fever, generalized morbilliform rash, leukocytosis with marked eosinophilia, and hepatic dysfunction 3 weeks after starting allopurinol therapy (300 mg/day for 3 days followed by 200 mg/day) for hyperuricemia and arthritis. The clinical findings were judged to be a probable drug reaction according to the Naranjo probability scale. The drug-induced hypersensitivity syndrome (DHS) resolved after withdrawal of allopurinol and initiation of systemic corticosteroid therapy. However, there was progressive worsening of anemia with reticulocytopenia; PRCA was suspected. PRCA was judged to be a possible drug reaction according to the Naranjo probability scale. The patient refused blood transfusion and bone marrow biopsy. Recombinant human erythropoietin was initiated in addition to prednisolone 15 mg daily. Eleven days later (approximately 7 wk after allopurinol withdrawal), both the hemoglobin level and reticulocyte count began to rise. The patient consented to a bone marrow study at that time, which confirmed the presence of dysplasia involving only the erythroid lineage.
Allopurinol may induce DHS, aplastic anemia, and, in rare instances, PRCA. We report the first case of PRCA concurrent with allopurinol-induced DHS in a patient with chronic kidney disease. Discontinuation of allopurinol is the first step in the treatment of such cases. The slow recovery of PRCA might be partly attributed to her underlying chronic kidney disease.
To minimize serious DHS, proper indications for treatment and dosage adjustment should be closely observed when starting allopurinol therapy in patients with chronic kidney disease.
报告1例别嘌醇治疗后发生的罕见的合并超敏反应综合征和纯红细胞再生障碍性贫血(PRCA)病例。
一名患有系膜增生性肾小球肾炎的43岁女性,在开始使用别嘌醇治疗(第1天300mg,共3天,之后每天200mg)高尿酸血症和关节炎3周后,出现发热、全身性麻疹样皮疹、白细胞增多伴明显嗜酸性粒细胞增多以及肝功能障碍。根据Naranjo概率量表,临床发现被判定为可能的药物反应。停用别嘌醇并开始全身使用糖皮质激素治疗后,药物性超敏反应综合征(DHS)得到缓解。然而,贫血进行性加重伴网织红细胞减少;怀疑为PRCA。根据Naranjo概率量表,PRCA被判定为可能的药物反应。患者拒绝输血和骨髓活检。除每天15mg泼尼松龙外,开始使用重组人促红细胞生成素。11天后(别嘌醇停药后约7周),血红蛋白水平和网织红细胞计数均开始上升。此时患者同意进行骨髓检查,结果证实仅红系存在发育异常。
别嘌醇可能诱发DHS、再生障碍性贫血,在罕见情况下还可诱发PRCA。我们报告了首例慢性肾脏病患者中PRCA与别嘌醇诱发的DHS同时发生的病例。停用别嘌醇是此类病例治疗的第一步。PRCA恢复缓慢可能部分归因于其潜在的慢性肾脏病。
为使严重的DHS风险降至最低,在慢性肾脏病患者开始使用别嘌醇治疗时,应密切观察治疗的适当指征和剂量调整。