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两例肌原纤维肌病:遗传学与生活质量研究

Two Cases of Myofibrillar Myopathies: Genetic and Quality of Life Study.

作者信息

Angelini Corrado, Ceolin Chiara, Rodriguez Alicia Aurora, Nigro Vincenzo

机构信息

Neuromuscular Laboratory, Department of Neurosciences, University of Padova, Campus Biomedico Pietro d'Abano, 35131 Padua, Italy.

Geriatrics, University of Padova, 35128 Padua, Italy.

出版信息

Muscles. 2023 Apr 6;2(2):177-186. doi: 10.3390/muscles2020013.

Abstract

We describe two cases of myofibrillar myopathies, due to different gene mutations. The first was a girl with cardiomyopathy and sensory axonal neuropathy that underwent cardiac transplantation at 15 years and suffers from rotatory scoliosis due to BAG3 mutation. The second is a male patient, with evident limb-girdle weakness since age 3. Two muscle biopsies were performed at ages 3 and 15, with muscle MRI, and LDB3 gene sequence analysis also carried out. Muscle biopsies revealed the presence of dystrophic changes in the first biopsy and myopathic abnormalities in the second, and the MRI images of the lower limbs showed an asymmetrical involvement in the thigh of quadriceps muscles and in the calf of gastrocnemius muscles. The patient was responsive to treatment with an intermittent steroid regimen and muscle-strengthening exercises. Considerations on both muscle-bone interaction and psychological and socioeconomic conditions are carried out for both cases.

摘要

我们描述了两例由不同基因突变引起的肌原纤维肌病病例。第一例是一名患有心肌病和感觉轴索性神经病的女孩,她在15岁时接受了心脏移植,因BAG3基因突变而患有旋转性脊柱侧弯。第二例是一名男性患者,自3岁起就有明显的肢带肌无力。分别在3岁和15岁时进行了两次肌肉活检,并进行了肌肉MRI检查以及LDB3基因序列分析。肌肉活检显示,第一次活检存在营养不良性改变,第二次活检存在肌病异常,下肢MRI图像显示股四头肌大腿和腓肠肌小腿存在不对称受累。该患者对间歇性类固醇治疗方案和肌肉强化锻炼有反应。对这两个病例都进行了关于肌肉与骨骼相互作用以及心理和社会经济状况的考量。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2a21/12225336/200833289fe9/muscles-02-00013-g002.jpg

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