Marozzo Roberta, Pegoraro Valentina, Angelini Corrado
IRCCS San Camillo Hospital, 30126 Venice, Italy.
Diagnostics (Basel). 2020 Sep 18;10(9):713. doi: 10.3390/diagnostics10090713.
Becker muscular dystrophy (BMD) is an X-linked recessive disorder caused by dystrophin gene mutations. The phenotype and evolution of this muscle disorder are extremely clinical variable. In the last years, circulating biomarkers have acquired remarkable importance in their use as noninvasive biological indicators of prognosis and in monitoring muscle disease progression, especially when associated to muscle MRI imaging. We investigated the levels of circulating microRNAs (myo-miRNAs and inflammatory miRNAs) and of the proteins follistatin (FSTN) and myostatin (GDF-8) and compared results with clinical and radiological imaging data. In eight BMD patients, including two cases with evolving lower extremity weakness treated with deflazacort, we evaluated the expression level of 4 myo-miRNAs (miR-1, miR-206, miR-133a, and miR-133b), 3 inflammatory miRNAs (miR-146b, miR-155, and miR-221), FSTN, and GDF-8 proteins. In the two treated cases, there was pronounced posterior thigh and leg fibrofatty replacement assessed by muscle MRI by Mercuri score. The muscle-specific miR-206 was increased in all patients, and inflammatory miR-221 and miR-146b were variably elevated. A significant difference in myostatin expression was observed between steroid-treated and untreated patients. This study suggests that microRNAs and myostatin protein levels could be used to better understand the progression and management of the disease.
贝克肌营养不良症(BMD)是一种由肌营养不良蛋白基因突变引起的X连锁隐性疾病。这种肌肉疾病的表型和病程在临床上具有极大的变异性。近年来,循环生物标志物作为预后的非侵入性生物学指标以及监测肌肉疾病进展方面已变得极为重要,尤其是与肌肉磁共振成像(MRI)相关联时。我们研究了循环微RNA(肌源性微RNA和炎性微RNA)以及卵泡抑素(FSTN)和肌肉生长抑制素(GDF - 8)蛋白的水平,并将结果与临床和放射影像学数据进行了比较。在8例BMD患者中,包括2例接受地夫可特治疗且下肢无力症状仍在进展的患者,我们评估了4种肌源性微RNA(miR - 1、miR - 206、miR - 133a和miR - 133b)、3种炎性微RNA(miR - 146b、miR - 155和miR - 221)、FSTN和GDF - 8蛋白的表达水平。在2例接受治疗的患者中,通过Mercuri评分的肌肉MRI评估显示大腿后部和小腿有明显的纤维脂肪替代。所有患者的肌肉特异性miR - 206均升高,炎性miR - 221和miR - 146b也有不同程度的升高。在接受类固醇治疗和未接受治疗的患者之间,观察到肌肉生长抑制素表达存在显著差异。这项研究表明,微RNA和肌肉生长抑制素蛋白水平可用于更好地了解该疾病的进展和治疗。