Chongqing Key Laboratory of Neurology, Department of Neurology, The First Affiliated Hospital of Chongqing Medical University, 1st Youyi Road, Yuzhong District, Chongqing, 400016, China.
Department of Geriatrics, The First Affiliated Hospital of Chongqing Medical University, 1st Youyi Road, Yuzhong District, Chongqing, 400016, China.
Acta Neurol Belg. 2022 Aug;122(4):969-977. doi: 10.1007/s13760-022-01974-5. Epub 2022 May 26.
This study aims to analyse the body composition features and its changes after treatment in patients with late-onset multiple acyl-CoA dehydrogenase deficiency (MADD).
Body composition was measured in patients with late-onset MADD, inflammatory myopathies, mitochondrial myopathy, and healthy controls. The correlation analyses between body composition and traditional parameters were performed. Comparisons between groups and receiver operating characteristic curve analyses were performed.
A total of 42 participants included 13 patients with late-onset MADD, 13 healthy controls, 10 with inflammatory myopathy, and 6 with mitochondrial myopathy. Bilateral grip strength and forced vital capacity (FVC) were moderate-strong correlated with skeletal muscle mass (right hand grip strength: r = 0.728, P < 0.001; left hand grip strength: r = 0.676, P < 0.001; FVC: r = 0.754, P < 0.001). Serum CK was moderately and negatively correlated with right hand grip strength (r = - 0.618, P = 0.005), left hand grip strength (r = - 0.630, P = 0.004), FVC (r = - 0.665, P = 0.002), manual muscle testing (MMT) (r = - 0.729, P = 0.000), and lean body mass skeletal muscle percentage (r = - 0.501, P = 0.029). Body composition features in patients with late-onset MADD were as follows: (1) obvious fat accumulation, (2) reduction of muscle mass, and (3) reduction of body water and intracellular water ratio. Some indicators of body composition were found to be valuable in diagnosis and eliminating differential diagnoses, such as visceral fat area (sensitivity 84.62%; specificity 92.31%; AUC 0.905) and fat mass (sensitivity 84.62%; specificity 75.00%; AUC 0.837). Seven patients were followed-up (2-9 months). Prior to treatment, the changes in body composition in these patients were conflicting.
Hand grip strength and FVC were strongly associated with body composition. Body composition features in late-onset MADD are fat accumulation, muscle loss, decrease in total body water, and intracellular water ratio. Body composition features are valuable for diagnosis and assessment.
本研究旨在分析迟发性多酰基辅酶 A 脱氢酶缺乏症(MADD)患者治疗后的身体成分特征及其变化。
对迟发性 MADD、炎症性肌病、线粒体肌病患者及健康对照者进行身体成分测量。对身体成分与传统参数进行相关性分析。组间比较及受试者工作特征曲线分析。
共纳入 42 名参与者,包括 13 例迟发性 MADD 患者、13 例健康对照者、10 例炎症性肌病患者和 6 例线粒体肌病患者。双侧握力和用力肺活量(FVC)与骨骼肌质量呈中高度相关(右手握力:r=0.728,P<0.001;左手握力:r=0.676,P<0.001;FVC:r=0.754,P<0.001)。血清肌酸激酶(CK)与右手握力(r=-0.618,P=0.005)、左手握力(r=-0.630,P=0.004)、FVC(r=-0.665,P=0.002)、徒手肌力测试(MMT)(r=-0.729,P=0.000)和瘦体重骨骼肌百分比(r=-0.501,P=0.029)呈中度负相关。迟发性 MADD 患者的身体成分特征如下:(1)明显的脂肪堆积;(2)肌肉量减少;(3)身体水分和细胞内水分比例减少。一些身体成分指标在诊断和排除鉴别诊断方面具有一定价值,如内脏脂肪面积(敏感性 84.62%;特异性 92.31%;AUC 0.905)和脂肪量(敏感性 84.62%;特异性 75.00%;AUC 0.837)。对 7 例患者进行了随访(2-9 个月)。治疗前,这些患者的身体成分变化存在矛盾。
握力和 FVC 与身体成分密切相关。迟发性 MADD 的身体成分特征为脂肪堆积、肌肉减少、全身总水量和细胞内水分比例降低。身体成分特征对诊断和评估具有重要价值。