Park J H, Niermann K J, Ryder N M, Nelson A E, Das A, Lawton A R, Hernanz-Schulman M, Olsen N J
Vanderbilt University Medical School, Nashville, Tennessee 37232, USA.
Arthritis Rheum. 2000 Oct;43(10):2359-67. doi: 10.1002/1529-0131(200010)43:10<2359::AID-ANR25>3.0.CO;2-D.
To characterize metabolic abnormalities in the muscles of children with the juvenile variant of dermatomyositis (JDM) by the use of noninvasive P-31 magnetic resonance spectroscopy (MRS).
Thirteen patients with JDM (ages 4-16 years) were studied. Biochemical status was evaluated with P-31 MRS by determining the concentrations of the high-energy phosphate compounds, ATP and phosphocreatine (PCr), ratios of inorganic phosphate (Pi) to PCr (Pi:PCr ratio), levels of free cytosolic ADP, and phosphorylation potentials (PPs) during rest, exercise, and recovery.
Significant metabolic abnormalities were observed in the thigh muscles of 10 severely affected patients during rest, 2 graded levels of exercise, and recovery. Mean ATP and PCr levels in the muscles of JDM patients were 35-40% below the normal control values (P < 0.003). These data, along with elevated Pi:PCr ratios, higher ADP levels, and abnormal values for PPs, indicated defective oxidative phosphorylation in the mitochondria of diseased JDM muscles. MRS findings were normal in 2 additional patients who had improved with prednisone treatment and in 1 patient who had no muscle weakness (amyopathic variant of JDM).
JDM patients can be monitored with noninvasive P-31 MRS without sedation. Biochemical defects in energy metabolism are concordant with the weakness and fatigue reported by JDM patients. Quantitative MRS data are useful for evaluating patients and optimizing drug treatment regimens.
通过使用无创³¹P磁共振波谱(MRS)来描述青少年型皮肌炎(JDM)患儿肌肉中的代谢异常情况。
对13例JDM患者(年龄4 - 16岁)进行研究。通过³¹P MRS测定高能磷酸化合物、三磷酸腺苷(ATP)和磷酸肌酸(PCr)的浓度、无机磷酸(Pi)与PCr的比值(Pi:PCr比值)、游离胞质二磷酸腺苷(ADP)水平以及静息、运动和恢复过程中的磷酸化电位(PPs),以此评估生化状态。
在10例严重受累患者的大腿肌肉静息、2级运动和恢复过程中观察到明显的代谢异常。JDM患者肌肉中的平均ATP和PCr水平比正常对照值低35 - 40%(P < 0.003)。这些数据,连同升高的Pi:PCr比值、更高的ADP水平以及PPs的异常值,表明患病的JDM肌肉线粒体中的氧化磷酸化存在缺陷。另外2例经泼尼松治疗后病情改善的患者以及1例无肌无力的患者(JDM无肌病型)的MRS结果正常。
JDM患者可在无需镇静的情况下通过无创³¹P MRS进行监测。能量代谢中的生化缺陷与JDM患者报告的肌无力和疲劳情况一致。定量MRS数据有助于评估患者并优化药物治疗方案。