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秋水仙碱诱发的青少年家族性地中海热肌病。

Colchicine-induced myopathy in a teenager with familial Mediterranean fever.

作者信息

Sayarlioglu Mehmet, Sayarlioglu Hayriye, Ozen Suleyman, Erkoc Reha, Gul Ahmet

机构信息

Department of Internal Medicine, Division of Rheumatology, Medical Faculty, Yuzuncu Yil University, Van, Turkey.

出版信息

Ann Pharmacother. 2003 Dec;37(12):1821-4. doi: 10.1345/aph.1D188.

Abstract

OBJECTIVE

To report a case of colchicine-induced myopathy in a teenager with familial Mediterranean fever (FMF).

CASE SUMMARY

A 15-year-old boy of Turkish origin, diagnosed as having FMF at the age of 14 years, was treated with colchicine 1.5 mg/d. He had experienced only 2 mild peritonitis attacks with fever within 1 year. The patient used the recommended dose regularly, and he described progressive proximal muscle weakness and generalized myalgias, which started 1 month before presentation. Physical examination showed proximal muscle weakness in his arms and legs. Laboratory tests revealed elevated serum creatine kinase, aspartate aminotransferase, alanine aminotransferase, and lactate dehydrogenase. All other laboratory values were within normal range. Electromyographic investigation revealed a myopathic pattern in proximal muscles without any neuropathic changes. A biopsy of the deltoid muscle showed vacuolar degeneration of striated muscle fibers with no inflammatory findings.

DISCUSSION

Colchicine, the most important drug in treatment of FMF, can cause myopathy in patients with impaired renal and hepatic function. In our patient, an objective causality scale showed that therapeutic doses of colchicine for FMF were the definite cause of myopathy, even though his renal and hepatic function were normal. The treatment of FMF attacks in patients who cannot use colchicine is an important problem. There are insufficient data about the use of immunosuppressive agents in the treatment of FMF attacks; however, we now successfully control the attacks with colchicine 0.5 mg/d and azathioprine 2 mg/kg/d.

CONCLUSIONS

Colchicine-induced myopathy should be excluded in patients with FMF who present with generalized muscle weakness. Clinicians should be aware that myopathy can occur in patients with FMF who have normal renal and hepatic function.

摘要

目的

报告1例患有家族性地中海热(FMF)的青少年因秋水仙碱诱发肌病的病例。

病例摘要

一名15岁土耳其裔男孩,14岁时被诊断为FMF,接受1.5mg/d秋水仙碱治疗。1年内他仅经历过2次伴有发热的轻度腹膜炎发作。患者规律使用推荐剂量,自述在就诊前1个月开始出现进行性近端肌无力和全身肌痛。体格检查显示其上肢和下肢近端肌无力。实验室检查显示血清肌酸激酶、天冬氨酸转氨酶、丙氨酸转氨酶和乳酸脱氢酶升高。所有其他实验室值均在正常范围内。肌电图检查显示近端肌肉呈肌病模式,无任何神经病变。三角肌活检显示横纹肌纤维有空泡变性,无炎症表现。

讨论

秋水仙碱是治疗FMF的最重要药物,可导致肾功能和肝功能受损的患者发生肌病。在我们的患者中,客观因果关系量表显示,尽管其肾功能和肝功能正常,但FMF治疗剂量的秋水仙碱是肌病的明确病因。对于不能使用秋水仙碱的患者,FMF发作的治疗是一个重要问题。关于使用免疫抑制剂治疗FMF发作的数据不足;然而,我们现在用0.5mg/d秋水仙碱和2mg/kg/d硫唑嘌呤成功控制了发作。

结论

出现全身肌无力的FMF患者应排除秋水仙碱诱发的肌病。临床医生应意识到,肾功能和肝功能正常的FMF患者也可能发生肌病。

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