Semi-Automated Analysis of Diaphragmatic Motion with Dynamic Magnetic Resonance Imaging in Healthy Controls and Non-Ambulant Subjects with Duchenne Muscular Dystrophy.
Semi-Automated Analysis of Diaphragmatic Motion with Dynamic Magnetic Resonance Imaging in Healthy Controls and Non-Ambulant Subjects with Duchenne Muscular Dystrophy.
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DOI：
10.3389/fneur.2018.00009
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发表时间：
2018
影响因子：
3.4
通讯作者：
Janiczek RL
中科院分区：
文献类型：
--
作者：
Bishop CA;Ricotti V;Sinclair CDJ;Evans MRB;Butler JW;Morrow JM;Hanna MG;Matthews PM;Yousry TA;Muntoni F;Thornton JS;Newbould RD;Janiczek RL
杜氏肌营养不良症（DMD）受试者会出现进行性肌肉损伤，导致横隔膜无力，最终需要通气。新兴的治疗方法已经引起了人们对更好地描述DMD呼吸障碍的自然史和对治疗的反应的兴趣。动态（电影）磁共振成像（MRI）可以提供一个更敏感的测量膈肌功能的DMD比常用的肺功能。本研究提出了一个分析管道，测量参数的动态MRI和它的应用程序，以调查健康对照组和非流动DMD男孩的呼吸功能的MRI措施。我们在基线时扫描了13名非活动DMD男孩和10名年龄匹配的健康男性志愿者，并在3、6和12个月后对DMD受试者的一个子集（n = 10、10、8）进行了评估。记录肺活量测定衍生的指标，包括用力肺活量。MRI衍生的测量包括肺横截面积（CSA）、矢状成像平面中的前、中和后肺长度以及动态MRI时间过程中的膈肌长度。回归分析表明，肺CSA和呼吸周期内的长度测量之间存在强线性相关性，DMD中这些相关性降低，并且DMD中的呼吸运动对改变肺容量的贡献效率较低。DMD患者的肺功能MRI测量值降低，控制了两组之间的身高差异：在最大吸气时，最大CSA和膈肌运动总距离分别小45%和37%。肺功能的MRI测量与肺功能测定数据相关，并显示与疾病进展的年龄和卧床月数的关系，表明它们提供了有临床意义的信息。12个月内MRI测量值的变化与膈肌和肋间肌减弱和进行性膈肌功能障碍一致。相比之下，在同一时期的传统肺量计测量中没有看到纵向变化。因此，动态MRI测量胸肌和肺功能被认为可以检测健康对照和DMD之间有意义的差异，并且可能对DMD非卧床男孩在相对较短的随访期内的功能变化敏感。
Subjects with Duchenne Muscular Dystrophy (DMD) suffer from progressive muscle damage leading to diaphragmatic weakness that ultimately requires ventilation. Emerging treatments have generated interest in better characterizing the natural history of respiratory impairment in DMD and responses to therapy. Dynamic (cine) Magnetic Resonance Imaging (MRI) may provide a more sensitive measure of diaphragm function in DMD than the commonly used spirometry. This study presents an analysis pipeline for measuring parameters of diaphragmatic motion from dynamic MRI and its application to investigate MRI measures of respiratory function in both healthy controls and non-ambulant DMD boys. We scanned 13 non-ambulant DMD boys and 10 age-matched healthy male volunteers at baseline, with a subset (n = 10, 10, 8) of the DMD subjects also assessed 3, 6, and 12 months later. Spirometry-derived metrics including forced vital capacity were recorded. The MRI-derived measures included the lung cross-sectional area (CSA), the anterior, central, and posterior lung lengths in the sagittal imaging plane, and the diaphragm length over the time-course of the dynamic MRI. Regression analyses demonstrated strong linear correlations between lung CSA and the length measures over the respiratory cycle, with a reduction of these correlations in DMD, and diaphragmatic motions that contribute less efficiently to changing lung capacity in DMD. MRI measures of pulmonary function were reduced in DMD, controlling for height differences between the groups: at maximal inhalation, the maximum CSA and the total distance of motion of the diaphragm were 45% and 37% smaller. MRI measures of pulmonary function were correlated with spirometry data and showed relationships with disease progression surrogates of age and months non-ambulatory, suggesting that they provide clinically meaningful information. Changes in the MRI measures over 12 months were consistent with weakening of diaphragmatic and inter-costal muscles and progressive diaphragm dysfunction. In contrast, longitudinal changes were not seen in conventional spirometry measures during the same period. Dynamic MRI measures of thoracic muscle and pulmonary function are, therefore, believed to detect meaningful differences between healthy controls and DMD and may be sensitive to changes in function over relatively short periods of follow-up in non-ambulant boys with DMD.
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3.5
作者：
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通讯作者：
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